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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction [1 ed.]
 9781907826733, 9781907826146

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction Extra Resources The resources found on the cd that accompanies the print book can be downloaded for free from the book’s page on the J&R Press Website: www.jr-press.co.uk

© 2014 J&R Press Ltd All rights reserved. No part of this publication may be reproduced, stored in a retrieval system or transmitted in any form or by any means, electronic, mechanical, photocopying, recording, scanning or otherwise, except under the terms of the Copyright Designs and Patents Act 1988 or under the terms of a licence issued by the Copyright Licensing Agency Ltd, without the permission in writing of the Publisher. Requests to the Publisher should be addressed to J&R Press Ltd, Farley Heath Cottage, Albury, Guildford GU5 9EW, or emailed to [email protected]. The use of general descriptive names, registered names, trademarks, etc. in this publication does not imply, even in the absence of a specific statement, that such names are exempt from the relevant protective laws and regulations and therefore free for general use. Library of Congress Cataloguing in Publication Data British Library Cataloguing in Publication Data A catalogue record for this book is available from the British Library Copyediting: Karen Weller, www.scribe-editorial.com Project management, typesetting and design: J&R Publishing Services Ltd, Guildford, Surrey, UK; www.jr-publishingservices.co.uk

Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction The Spires Cleft Centre SLT Team Ginette Phippen (Editor)

J&R Press Ltd

Image permissions Chapter 3.2 and Resources section GOS.SP.ASS form Sell, D., Harding, A. and Grunwell, P. (1999). GOSSPASS’98: an assessment for speech disorders associated with cleft palate and/or velopharyngeal dysfunction (revised). International Journal of Language and Communication Disorders, 34(1), 17-33. Reproduced with the authors’ permission. Chapter 4 Figure 4.5 Palatal Lift Figure 4.6 Speech Bulb Reproduced by kind permission of Elsevier. Originally published in Peterson-Falzone et al., 2010, Cleft Palate Speech (4th ed.) pp358-359. Copyright Elsevier. Chapter 8.1 & all Boardmaker™ images in Resources section Figure 8.2 Hold nose prompt card (Boardmaker™) The Picture Communication Symbols ©1981–2010 by Mayer-Johnson LLC. All Rights Reserved Worldwide. Used with permission. Boardmaker™ is a trademark of Mayer-Johnson LLC. DynaVox Mayer-Johnson 2100 Wharton Street Suite 400 Pittsburgh, PA 15203 Phone: 1 (800) 588-4548 Fax: 1 (866) 585-6260 Email: [email protected] Web site: www.mayer-johnson.com Resources section: Place map for high pressure consonants Image reproduced by kind permission of Elsevier. Originally published in Peterson-Falzone, Trost-Cardamone, Karnell, Hardin-Jones, 2006 The Clinician’s Guide to Treating Cleft Palate Speech. P134. Copyright Elsevier.

Contents Acknowledgements About the contributors Foreword by Stephen Robinson Preface

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PART ONE: Setting the Scene 1 Introduction to Cleft Palate Ginette Phippen

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2 Velopharyngeal Dysfunction Carrie Luscombe

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PART TWO: From Identification to Intervention 3 Assessment Section 1: Oral Examination Fiona Jeyes Section 2: Speech Ginette Phippen and Sandra Treslove 4 Assessment and Management of Velopharyngeal Dysfunction (VPD) Leda Koutsoulieri 5 Hearing Difficulties Lucy McAndrew

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59 85

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Contents

6 Voice Issues Leda Koutsoulieri

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7 Principles of Early Intervention Sandra Treslove

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8 Speech Sound Therapy Helen Piggott

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9 Applying the Psycholinguistic Framework in Cleft Speech Therapy Benedicta Isaac-Kumar 10 Therapy approaches Section 1: Oral Motor Therapy: Is there a place for it in speech therapy for children with cleft palate? Benedicta Isaac-Kumar

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Section 2: Active Nasal Fricative Helen Piggott and Fiona Jeyes

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Section 3: Electropalatography Lucy McAndrew (with therapy for EPG by Helen Piggott)

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Section 4: Speech Therapy and Other Non-Surgical Treatments for Nasal Speech Ginette Phippen

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PART THREE: Working in Partnership 11 Working in Partnership Catherine Catterall and Sandra Treslove

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Contents

12 Working with Children with Complex Needs: Meeting the challenges Zoe Gordon and Carrie Luscombe

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297

13 Understanding 22q11 Deletion Syndrome Carrie Luscombe

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14 Section 1: Supporting Feeding in the Child with a Cleft Section 2: Feeding Difficulties in 22q11 DS Zoe Gordon

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Glossary Index

397 399

Acknowledgements Acknowledgements are due to: • The children and young people with cleft palate and their families who have allowed us to feature them. We hope that this book will effectively support those who are supporting them. • Those who provided us with their thoughts and ‘speech bubbles’ – thank you all. • Cindy Williams, speech and language therapy assistant, for her artwork and days, nights and weekends spent getting the whole thing together. • Rachael and Jim Wilkie at J & R Press. • Colleagues at the Spires Cleft Centre in Salisbury and Oxford (some of whose contributions go uncredited). • Colleagues in the Salisbury SLT department for encouragement and moral support. • Simon Spencer for additional artwork. • Steve Clark and Mike Osborn for producing and arranging the music on our CD. • Jo Laing at SDH library for literature searching. • Salisbury NHS Foundation Trust and Oxford Health NHS Foundation Trust for supporting the project. A final thank you to families and friends for support and encouragement throughout.

About the contributors Catherine Catterall Cert. MRCSLT; BMedSci (Hons) Speech Science, University of Sheffield, 2001. Catherine qualified as a Speech and Language Therapist in 2001. She has experience of working within a large community health trust, supporting children with a wide range of speech and language difficulties in community clinics and a variety of educational settings. Since 2009 she has worked as a cleft link SLT, and has recently completed the first year of the MSc (Speech and Cleft) through distance learning with the University of Sheffield. Catherine joined the Spires Cleft Team at Salisbury in Spring 2013.

Zoe Gordon Cert. MRCSLT; BSc (Hons) Speech and Language Therapy, University of Wales Institute, Cardiff, 1999. After eighteen months in Zoe’s first SLT post, a passion for travel led her overseas to a mid-west America speech therapy camp, Asia, and eventually the southern hemisphere where she worked in New Zealand for four years. It was here that she met Maeve, an inspiring cleft palate specialist SLT and her passion for working with children with craniofacial conditions and feeding difficulties was born. Zoe joined the Oxford cleft-craniofacial teams in 2005 and has now put her passport to one side. Her current area of interest is researching feeding difficulties in infants with complex craniofacial syndromes.

Benedicta Isaac-Kumar Benedicta trained as a speech and language therapist and audiologist in 1997 followed by a post-graduate degree in psychology and certificate in cleft palate studies. Her passion lay in the areas of children with speech difficulties and children with social communication difficulties. Working at the Spires Cleft Centre gave her ample opportunity to explore research and work in the area of childhood speech impairments. Benedicta has founded and heads the charity, Disabled Child-India, which focuses on transferring knowledge and skills from countries like the UK, to India, in the area of special needs. She has also

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established a learning centre for children with autism in Tirunelvelli, South India, and is now developing her interest in the area of social communication disorders.

Fiona Jeyes Cert. MRCSLT; Post Graduate Certificate in Cleft Palate Studies, University of Sheffield. Fiona worked in a wide range of community and educational settings for many years before joining the Spires Cleft Palate service in Salisbury. She is keen to encourage links between hospital and home SLT services through joint working, liaison and informal teaching sessions. Fiona is responsible for the Spires SLT training programme, aiming to increase therapists’ knowledge and confidence about this client group in order to help them to target their interventions.

Leda Koutsoulieri Cert. MRCSLT; BA Psychology; BSc (Hons) Clinical Communication Studies, City University, London; MSc Speech and Cleft, University of Sheffield. Leda works as a Specialist Speech and Language Therapist in the Spires Cleft Centre, at the John Radcliffe Hospital, Oxford. Previously she worked for the Cleft Lip and Palate Unit, Great Ormond Street Hospital NHS Trust, and prior to this as a paediatric therapist in Ealing. Her clinical interests include voice disorders, and the assessment and management of developmental verbal dyspraxia in cleft palate/velopharyngeal dysfunction. She currently specialises in prosthetic intervention for the management of VPD. She provides regular training to speech and language therapists.

Carrie Luscombe Cert. MRCSLT; BA (Hons) Linguistics and Language Pathology from Reading University in 2000; MSc Speech and Cleft, University of Sheffield. After qualifying, Carrie worked in a mixed adult and paediatric post in Essex. During her time there, Carrie worked as a link SLT for the Great Ormond Street Cleft Service based at St Andrew’s in Broomfield. This fuelled her aspiration to work in cleft full time and she came to work in Oxford in 2003. Carrie is the Principal Speech and Language Therapist with the Spires Cleft Centre and

About the contributors

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Oxford Craniofacial Unit, based at the John Radcliffe Hospital. Her clinical and research interests include assessment and management of velopharyngeal dysfunction, issues related to speech and quality of life and working with people with 22q 11 deletion syndrome and craniofacial syndromes.

Lucy McAndrew Cert. MRCSLT; BSc (Hons) Speech Sciences, University College London 1994; Postgraduate Certificate in Cleft Palate Studies, Sheffield; MSc Speech and Cleft (in progress). Lucy is a Specialist SLT at the Spires Cleft Centre, Oxford where she has been based for the last nine years. Prior to this she worked for several years as a paediatric therapist before specialising in cleft palate and velopharyngeal dysfunction, working with Anne Harding-Bell at Addenbrooke’s Hospital, Cambridge. Her clinical and research interests include ENT and hearing difficulties related to cleft palate and electropalatography.

Ginette Phippen Cert. MRCSLT; BSc Clinical Language Science, Leeds, 1989; Postgraduate Cleft Palate Studies, Homerton College, Cambridge, 2003; Doctorate in Clinical Practice, University of Southampton, 2013. Ginette is the Lead Speech and Language Therapist for the Spires Cleft Centre (Salisbury & Oxford) where she has worked for the last 10 years. Before that she worked for a number of years as specialist SLT with a community learning disability team for adults and children and in variety of other paediatric SLT roles in Gloucestershire. Ginette has just completed an exploratory study of visual feedback therapy for nasal speech as part of a Clinical Doctorate programme. Research is one her main areas of interest and she oversees the Spires SLT team research programme, with current involvement in a number of exciting national and international projects.

Helen Piggott Cert. MRCSLT; graduated from Birmingham with BSc (Hons) in 1985. Helen is a Specialist SLT with over 25 years of experience. She has worked for several NHS Trusts including Shropshire, Northants and Milton Keynes,

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working within community settings as well as supporting children with moderate learning difficulties. Helen initially became involved with cleft as a Link SLT before joining the Oxford Spires Team. Her specific area of interest is speech disorders in addition to CLP, with a focus on devising and implementing therapy programmes and supporting clinicians in the community.

Sandra Treslove Cert. MRCSLT; B.A. (Hons) Drama, Hull; MSc Speech and Language Therapy, City University; Post Graduate Certificate in Cleft Palate Studies, University of Sheffield. Sandra originally read Drama at Hull University. She worked as an actor-musician in community theatre for ten years before packing away the instruments and retraining as a speech and language therapist at City University. She undertook the Postgraduate Certificate in Cleft Lip and Palate Studies at Sheffield University, and has worked with the Spires Cleft Centre since 2006. Her clinical interests include early years intervention and voice disorders.

Foreword Modern cleft lip and palate care involves a core multidisciplinary team of specialist surgeons, nursing, psychology, orthodontics and speech and language therapists to provide an integrated approach to managing a complex problem. The care pathway starting before birth will often not be complete until early adult life. The main focus of early management of cleft palate concentrates on feeding and surgical repair before the end of the first year combined with monitoring speech and hearing development. Learning to speak is considered one of the most important parts of child development. A failure to hear and communicate effectively can influence education and long-term employment prospects. A cleft can have a significant impact on speech and early detection of speech difficulties is crucial if long-term problems are to be avoided. For a minority of individuals the speech deficit will require ongoing therapy for many years. This book sets out to explain the wide spectrum of speech therapy required to manage speech problems and velopharyngeal dysfunction (VPD) when associated with a cleft palate. It will also demonstrate how these skills cross over into the management of non-cleft VPD and other acquired anomalies in both children and adults. It has been written by the speech therapy team of the Spires Cleft Service and will appeal to clinicians working directly in the field and to educate the wider body of healthcare professionals. Stephen Robinson Clinical Director, The Spires Cleft Centre (Oxford & Salisbury) Chair, CLAPA (Wessex)

Just a few thoughts about speech therapy in the 1960s It’s Wednesday afternoon, time to go to the hospital. Father would drive us there until Mother passed her driving test in 1969, I was 9 then. Time off school, I would take a note to the teacher explaining that I would not be back in the afternoon for lessons. Upon reaching the hospital we would go into the waiting room where I would see other children with clefts, I thought I was the only one! There was no talking; we just sat in the waiting room quietly. My mother would talk to other mothers but I just waited. We would then be called by Mrs P, a fierce and formidable lady, with Dame Edna glasses and a bun in her hair. She was tall and I was frightened of her. She would go through all these words and sounds with me, ‘Ssssss’ were always difficult and she would give me similar words to say, ‘sing swing, swim’ or we would just keep saying the ‘S’ sound. We would be given homework to do. Mother would sit me down and we would practise various words and sentences from an exercise book. My mother has kept them for over 50 years and they would be marked by a red star or, if I had made progress well in Mrs P’s eyes, a silver or gold star. I don’t think I ever enjoyed doing speech therapy but I think it really helped me for later life. Mrs P’s strict approach was not what I wanted but maybe I needed it. Today I think there are better ways of doing speech therapy from what I have seen. The trouble with Mrs P’s approach was that it killed confidence and confidence is something in short supply when you are a youngster, especially a youngster with a cleft. John S PS I love to listen to spoken words; I love the challenge of a speech after dinner. I have been asked to speak at my friend’s and best man’s funeral. I do not find it easy but I like to speak as well as I can.

Editor’s Preface Ginette Phippen Speech, as a form of communication, can be seen as a core life skill. The World Health Organization defines life skill as the ability for adaptive and positive behaviour that enables an individual to deal effectively with the demands and challenges of everyday life. Communication is one of 10 life skills proposed by the WHO as being essential for the healthy development of children and adolescents. The inspiration for this book has come from two sources. First of all, a parent who was requesting regular therapy sessions for her 4-year-old son despite living over one hour’s drive from the cleft centre. They had seen a community SLT at a clinic close to their home and the mum described the therapist as ‘really lovely with Harry, but ...’. The ‘but’ was added because the community SLT had said she did not know about cleft palate. Unfortunately, this meant that the mum was not confident that the community SLT could provide the therapy that her child needed. The second source of inspiration has been the biannual clinical network training days that we offer as a speech therapy team to community and cleft link SLTs from across the Spires regional area. These stimulating days absolutely confirm for me the need for and value of partnership working between our teams. They also demonstrate that some community SLTs do know more about cleft palate than others but that they all know a lot about delivering therapy to children, often in constraining contexts. This book is written precisely for these SLTs, who we rely on to support the delivery of advice and therapy for individuals with cleft palate and VPD, across a wide geographical area. The book begins with an overview of cleft palate and VPD. The second section moves on to identification and intervention, including the essential components of assessment and then specifically in relation to VPD. This is followed by an exploration of hearing and voice issues related to cleft and VPD. The next chapter considers the evidence for early intervention, describing a model for delivering one specific modelling approach. Speech sound therapy is covered with a very practical emphasis

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and a chapter focusing on the application of a psycholinguistic approach. Several specific therapy approaches are then explored (oral placement therapy, active nasal fricatives, electropalatography, therapy for nasal speech) and the evidence base evaluated. The final section of the book discusses partnership working in relation to speech sound therapy and supporting children with complex needs, feeding difficulties and specific syndromes where cleft and VPD are a feature. The contributors to the book are all speech and language therapists specialising in the area of cleft palate and VPD. The reader will encounter a range of writing styles but the emphasis throughout is on achieving a balance between theory and current evidence combined with practical guidance and resources. Finally, the aim is also to consider the broader context of the lived experience of individuals with cleft/VPD-related communication difficulties. This is achieved with reference to the available literature and through the contributions of some individuals receiving speech therapy. John S starts by describing in the Foreword his experiences of speech therapy in the 1960s. The ‘speech bubbles’ included throughout contain the words and pictures of children, young people and their parents, who were invited to ‘say what they thought about coming to speech therapy’. Notwithstanding the likely bias of the contributions received this adds an important and illuminating dimension.

1 Introduction to Cleft Palate Ginette Phippen

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Aim of this chapter To provide a brief overview of cleft lip and palate, including incidence, cause, classification and timing of primary surgical treatment.

Incidence and cause In the UK, approximately one thousand babies are born each year with a cleft lip, cleft palate or combination of the two (CRANE database, 2009). A cleft literally means ‘a separation’ or ‘split’ (CLAPA, 2009) and can involve the lip on one or both sides as well as the palate. Worldwide, the birth prevalence ranges from 1 in 1000 to 2.69 in 1000 births (McLeod et al., 2004) making cleft lip and/or palate one of the most common congenital anomalies. Its causes are not entirely understood and are likely to be multifactorial (Watson, 2001). There is good evidence from studies of embryology and genetics of the heterogeneity of the cleft palate population. Spriestersbach et al. (1973) demonstrate that isolated cleft palate and cleft lip are distinct in origin and environmental factors are likely to be of significance only in relation to cleft palate. In this early comprehensive review of cleft palate, Spriestersbach reports evidence of both genetic factors (e.g., chromosome defects and mutant genes) and environmental factors (e.g., anti-emetic and anti-epileptic drugs, and folic acid deficiency) and also discusses the likelihood of interaction between the two in many cases. More recently, Stanier and Moore (2004) report on advances in identifying the genes and gene pathways associated with clefting. They acknowledge the difficulty in relating experimental studies to the complex human situation but conclude that there should be optimism about further pinpointing the contributory factors.

• Research

The Cleft Collective is a UK-based research programme, funded by the Healing Foundation, which aims to investigate the causes and impact of cleft lip and palate. The programme hopes to recruit up to 5000 children and families into clinical studies from 2012.

Cleft types In 80% of babies, a cleft of the palate will be present. Cleft lip and palate is

Introduction to Cleft Palate

No cleft

Incomplete unilateral cleft lip

Complete unilateral cleft lip

No palatal cleft

Bifid uvula

Cleft Palate

Complete bilateral cleft lip Complete unilateral cleft lip & palate Figure 1.1 Cleft types

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more common in males, whilst isolated cleft palate occurs more often in females (Stanier & Moore, 2004). Cleft lip and palate can occur in isolation or as part of a complex disorder, and is described as a feature in over 400 known syndromes (Mossey et al., 2009). See Figure 1.1 for cleft types.

Cleft surgery In the UK, a cleft lip is usually surgically repaired at around three months of age and a cleft palate at 6–9 months of age (Watson, 2001). The timing of this surgery continues to be the subject of international debate. Rohrich et al. (2000; 1996) discuss how the controversy centres on the apparent conflict between a need for early palate repair for optimum speech development versus delayed palate repair to allow for undisturbed facial growth. Supporters of early palate repair (at 12 months of age and under) argue that the nature of speech development as a learned behaviour indicates that anatomy should be restored as soon as possible (Russell & Grunwell, 1993; Estrem & Broen, 1989).

• Research In an attempt to resolve this question a randomized controlled trial is currently under way involving cleft centres in the UK, Scandinavia and Brazil (Timing of Primary Surgery for Cleft Palate [TOPS], in progress). This study is aiming to determine whether surgery for cleft palate, using a specified technique, at age 6 months, when compared to surgery using the same technique at age 12 months, affects speech development outcomes at ages 3 and 5 years. Notwithstanding timing of primary surgery, cleft palate is a structural defect and it is therefore hoped that the initial surgical palate repair will result in adequate soft palate function, so that during speech air flows through the mouth rather than the nose. If this does not happen then air will escape into the nose and the child’s developing speech may have a nasal tone. This may be because the repaired soft palate is too short, or does not move well, or there is a residual hole in the palate known as a fistula (Mercer & Pigott, 2001). At least 20% of children will continue to experience escape of air down the nose after the primary cleft repair, resulting in nasal speech (Witt et al., 1997).

Introduction to Cleft Palate

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Burden of care Following primary surgery in infancy, in the UK, a child born with a cleft will be offered follow-up by the specialist multidisciplinary team into adolescence and sometimes adulthood. As the child grows up, further surgery may be required to improve speech, as well as to repair the residual bony defect in children born with a cleft affecting the alveolus (gum). Later still, when the child reaches teenage years, there may be discussions about surgical treatment to improve the appearance of the nose (rhinoplasty) and jaw/facial profile (osteotomy). Finally, over and above the surgical interventions, there will be multiple appointments with ENT (Ear, Nose and Throat), audiology, speech and language therapy, orthodontics, psychology and possibly others, combining to represent a significant burden of care for the child and family. g Further Reading: Watson A.C.H., Sell D.A. & Grunwell P. (2001) (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers.

t Useful Links: Cleft Lip and Palate Association (CLAPA) at http://www.clapa.com CLEFT COLLECTIVE at http://www.cleftcollective.org.uk

References CLAPA (2009). Understanding cleft lip and palate [online]. Available at: http://www.clapa. com/medical/cleft_lip_article/107/ CRANE database (2009). Annual Report. Available at: https://www.crane-database.org. uk. Estrem, T. & Broen, P. (1989). Early speech production of children with cleft palate. Journal of Speech, Language, and Hearing Research, 32, 12­–23. McLeod, N.M.H., Arana-Urioste, M.L. & Saeed, N.R. (2004). Birth prevalence of cleft lip and palate in Sucre, Bolivia. Cleft Palate-Craniofacial Journal, 41(2), 195–198. Mercer, N.S.G. & Pigott, R.W. (2001). Assessment and surgical management of velopharyngeal dysfunction. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers.

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Mossey, P.A. et al. (2009). Cleft lip and palate. The Lancet, 374(9703), 1773­–1785. Rohrich, R.J., Rowsell, A.R., Johns, D.F., Drury, M.A., Grieg, G., Watson, D.J. & Poole, M.D. (1996). Timing of hard palatal closure: A critical long-term analysis. Plastic and Reconstructive Surgery, 98(2), 236. Rohrich, R.J., Love, E. J., Byrd, H.S. & Johns, D.F. (2000). Optimal timing of cleft palate closure. Plastic and Reconstructive Surgery, 106(2), 413–422. Russell, J. & Grunwell, P. (1993). Speech development in children with cleft lip and palate. In P. Grunwell (Ed.), Analysing Cleft Palate Speech (pp.6–18). London: Whurr Publishers. Spriestersbach, D.C., Dickson, D.R., Fraser, F.C., Horowitz, S.L., McWilliams, B.J., Paradise, J.L. & Randall, P. (1973). Clinical research in cleft lip and cleft palate: The state of the art. The Cleft Palate Journal, 10, 113. Stanier, P. & Moore, G.E. (2004). Genetics of cleft lip and palate: Syndromic genes contribute to the incidence of non-syndromic clefts. Human Molecular Genetics, 13(s1), R73–R81. Watson, A.C.H. (2001). Embryology, aetiology and incidence. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Witt, P.D. et al. (1997). Surgical management of velopharyngeal dysfunction: Outcome analysis of autogenous posterior pharyngeal wall augmentation. Plastic & Reconstructive Surgery, 99(5), pp.1287–1296; discussion 1297–1300.

2 Velopharyngeal Dysfunction Carrie Luscombe

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Aim of this chapter This chapter will define the term velopharyngeal dysfunction (VPD) and its relationship to cleft palate and other conditions affecting palate function. It outlines the speech features associated with VPD, along with other sequelae of the condition, and concludes with a discussion of the impact of VPD on quality of life.

What is velopharyngeal dysfunction (VPD)? ‘Velo’ refers to the velum (the soft palate) and ‘pharyngeal’ refers to the pharynx (the throat). Together, the term is used to describe the palate to pharyngeal closure mechanism that operates in speech and swallowing. When the mechanism is deficient or inadequate, there is dysfunction. Various terms are used in the literature to describe impaired function of the velopharyngeal mechanism. Authors may use terms interchangeably or use one generic term to describe all types of velopharyngeal dysfunction of both cleft and non-cleft origin, for example, velopharyngeal inadequacy (VPI) or velopharyngeal dysfunction (VPD).

• Terminology

The following terminology was proposed by Trost-Cardamone (1989) to explain velopharyngeal disorders based on aetiology: Velopharyngeal Insufficiency can be either cleft or non-cleft in origin. Velopharyngeal insufficiency is impaired velopharyngeal function as a consequence of any structural defect of the palate or pharyngeal wall. Velopharyngeal Incompetence refers to a neuromotor or physiological disorder which results in poor movement of the velopharyngeal structures. This may be seen in patients with paresis, apraxia, and dysarthria. These conditions may be congenital or acquired. Velopharyngeal Mislearning includes sound specific/compensatory nasal emission, e.g., active nasal fricatives and velopharyngeal symptoms associated with hearing impairment. These are not caused by structural defects or neuromotor issues.

Velopharyngeal Dysfunction

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VPD is the generic term adopted throughout this book as it is in common usage in UK cleft centres.

What causes VPD? The cause of VPD may be structural, neurogenic or mechanical, or the result of mislearning as summarised in Table 2.1. In some individuals who present with VPD, there is a combination of aetiological factors, for example, cleft palate (structural) and apraxia (neurogenic). Cleft palate is the most common structural cause of VPD (Johns et al., 2003). Where VPD is the result of something other than cleft palate this is generally subsumed under the heading of non-cleft VPD. In the UK, this term is used to describe a problem with the structure or function of the soft palate in the absence of a diagnosis of cleft palate. This covers VPD resulting from conditions such as cerebral palsy, stroke, traumatic brain injury and a wide range of degenerative neurological conditions.

Cleft VPD The presence of an unrepaired palatal cleft, whether it is overt, submucous or an occult submucous cleft, is likely to result in some degree of VPD. This is due to the individual’s inability to valve air appropriately through the oral and nasal cavities to produce clear speech, prevented by the inadequate structure or function of the soft palate. Table 2.1 Causes of VPD Causes of VPD – Cleft and Non-Cleft Structural • Cleft palate • Short palate/Large nasopharynx • Post-adenoidectomy • Post-ablative surgery (e.g., cancer)

Mechanical • Enlarged tonsils • Posterior pillar webbing

Neurogenic • Cranial nerve damage/dysfunction • Dysarthria (muscle weakness) • Dyspraxia (muscle incoordination)

Mislearning • Speech sound error (active nasal fricative) • Hearing impairment

Adapted from Peterson-Falzone (2006)

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• Terminology

Overt cleft (cleft palate) – Clefts of the secondary (soft) palate range from minimal defects to complete clefts extending all the way forward to the region of the incisive foramen. Submucous cleft palate (SMCP) ­– A significant defect of the soft palate, without an actual opening into the nasal cavity. The three classic stigmata of a SMCP are: • A bifid uvula • A zona pellucida: this is where the lack of normal muscle of the soft palate can cause the lining of the mouth (mucosa) to look so thin that the soft palate appears a bluish colour on oral examination • A notch into the hard palate. Occult submucous cleft palate – A defect in the muscular bulk on the upper layer of the soft palate (on the nasal surface) that can only be seen using nasendoscopy. None of the above stigmata are present.

See Chapter 3 for further information on carrying out oral examination and identification of SMCP. Following primary repair of the cleft palate in infancy, around 20% of children will continue to experience symptoms of VPD (Witt et al., 1997). This may be because the repaired soft palate is too short, or does not move well, or there is a fistula (Mercer & Pigott, 2001). A fistula is a residual hole in the palate following palate repair. It can be difficult to know if VPD symptoms are related only to a fistula or also related to dysfunction of the soft palate. Isberg and Henningson (1985) reported that fistulae are causally related to VPD. They demonstrated that individuals showed better velopharyngeal movement when the fistula was covered than when it was left uncovered. A number of children born with a cleft palate may also go on to develop VPD as they grow, as a consequence of: • Adenoid involution, a normal developmental process where the adenoid tissue shrinks, occurring between 6 and 16 years of age (Mason et al., 1980)

Velopharyngeal Dysfunction

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• Changes to the dimensions of the velopharynx e.g., an increase in size due to growth. To date, research on the number of children affected by these changes has been inconclusive with adenoid involution likely to be one of a number of contributing factors, including palate length (Morris et al., 1990; Van Demark et al., 1988). With this in mind, it is essential that children born with a palatal cleft continue to have speech reviews as they grow, to monitor for increased symptoms of VPD. Occasionally, VPD will persist in children with cleft palate even when additional surgery to improve speech (secondary/speech surgery) has been carried out (Witt et al., 1998). See Chapter 4 for discussion of approaches to the assessment and management of persisting VPD.

Non-cleft VPD Non-cleft VPD may have a structural, mechanical or neurogenic cause, be related to velopharyngeal mislearning or be multifactorial (see Table 2.1). Differential diagnosis is often challenging for children presenting with noncleft VPD.

Structural and mechanical causes: Tonsils and adenoids The tonsils are usually found in the oral cavity between the anterior and posterior faucial pillars, as shown in Figure 2.1. The adenoids are situated on the posterior wall of the pharynx, behind the nasal cavities, at or just above the point at which the velum makes contact with the posterior pharyngeal wall during elevation and closure. They are not visible during oral examination. The adenoids increase in size in the childhood years, until around age 7 years and then begin to recede and involute (shrink), so that by the adult years they have usually disappeared (Mason et al., 1980).

• Terminology

The tonsils and adenoids comprise of lymphoid tissue which are involved in fighting bacterial and viral infections. Enlarged (hypertrophied) tonsils can impact on resonance and VP function. Sometimes otherwise healthy tonsils are enlarged or posteriorly displaced into the oropharynx, where they may have a negative effect on oral and nasal

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

resonance and on articulation, as described below (Henningson & Isberg, 1988; Shprintzen et al., 1987). One or more of the following speech symptoms may occur: • Abnormal oral resonance, often referred to as ‘potato in the mouth’ resonance. This is a distortion in oral resonance caused by the tonsils sitting in the pharynx, which dampens the sound that would normally resonate in a larger cavity resulting in a ‘muffled’ voice quality. • Hypernasality and/or nasal emission. The upper poles of the tonsils may be interposed between the velum and the posterior pharyngeal wall preventing complete closure. The resulting VPD can be mild and/or inconsistent but may indicate the need for tonsillectomy. • Hyponasality. The tonsils may physically reduce nasal resonance by obstructing the postnasal space. • Vowel distortions. As vowels rely heavily on oral resonance, enlarged tonsils can alter this and affect vowel quality. • Consonant distortions. Tongue protrusion is often seen in individuals with enlarged tonsils as a strategy for opening up the airway.

Figure 2.1 Oral cavity, showing position of the tonsils

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Enlarged (hypertrophied) adenoids can make speech hyponasal (‘bunged up’) and this can be an indication for their surgical removal. A number of children then present with VPD following adenoidectomy, as a consequence of the sudden structural changes. The palate that used to close against the adenoidal pad now does not and the child fails to adjust to the increased velopharyngeal space (Witzel et al., 1986). The reported incidence of hypernasality postadenoidectomy varies greatly according to the literature, occurring between 1 in 1,500 and 1 in 10,000 procedures (Saunders et al., 2004). In some cases, adenoidectomy may reveal a pre-existing structural or neurogenic abnormality that is the cause of VPD (Witzel et al., 1986; Croft et al., 1981). There are also important non-speech symptoms in relation to tonsils and adenoids, which can be checked when taking a case history in order to decide whether an ENT referral or review is required: • Does the child snore? • Is the child a noisy breather? • Does the child ever wake at night and gasp for breath or stop breathing, i.e., signs of sleep apnoea? Are they frequently lethargic in the daytime? • Does the child have difficulty with chewing and swallowing? • Does the child frequently suffer from throat infections? • Does the child have middle ear problems?

Structural causes: Velopharyngeal disproportion This refers to a mismatch between the length of the velum (soft palate) and the depth of the pharynx, with or without signs of a submucous cleft palate. Possible contributing factors include a short hard palate, a short velum or increased pharyngeal depth. In addition to these congenital problems, adenoidectomy or tonsillectomy that results in an abnormally deep pharynx can also cause a problem. Similarly, surgery in this area to remove a malignant or non-malignant growth can also result in inadequate VP closure and symptoms of VPD. Abnormal pharyngeal depth is also seen in individuals with congenital abnormalities of the cervical spine, for example in Klippel-Feil Syndrome (Helmi & Pruzansky, 1980). If the posterior processes of two or more cervical vertebrae are fused, or if one or more vertebrae are displaced posteriorly, the effect is to pull the posterior pharyngeal wall back, creating an abnormally

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

deep pharyngeal space. Another cause of an abnormally deep pharynx is a flattened cranial base angle, which also has the effect of moving the posterior pharyngeal wall back.

Neurogenic causes: Paresis Paresis (impairment of movement) of the velum alone or together with oral or pharyngeal muscles has been reported as either a temporary (Walter et al., 2012) or persisting condition (Dworkin & Johns Donnell, 1980). In some cases there is a congenital cause such as cranial nerve defects, with the vagus nerve most often involved, for example, in individuals with 22q 11 Deletion Syndrome or Worster-Drought Syndrome (see Chapter 12). In others, it is due to acquired disease or trauma resulting in dysarthria and associated immobile or weak palate and inadequate VP function.

Dysarthria Duffy (1995) describes specific features of velopharyngeal function in relation to each of the dysarthrias, as earlier categorised by Darley, Aronson and Brown (1975; 1969a; 1969b). This is summarised below: • Flaccid dysarthria: Velopharyngeal dysfunction dependent on site damage. Hypernasality, imprecise consonants, nasal emission, short consonants. In unilateral involvement, the velum pulls towards the non-paralysed side on phonation. • Spastic dysarthria: Hypernasality, pressure consonants more severely involved than in other dysarthrias; gag possibly hyperactive, but velar movement may be slow or minimal on phonation. • Ataxic dysarthria: Abnormal resonance is rare. Infrequent episodes of hyponasality, presumably reflecting timing problems between velar and articulatory gestures. Oral examination is often normal. • Hypokinetic dysarthria: Increased nasal airflow on intended nonnasal consonants – slow velopharyngeal movements in speech. • Hyperkinetic dysarthria: Possible palatal pharyngeal myoclonus; possibly no effect on speech.

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• Unilateral upper motor neurone dysarthria: Accounts for hypernasality in 11% of the dysarthric patients seen at the Mayo Clinic. • Mixed dysarthria: Accounts for more than 34% of the dysarthric patients seen at the Mayo Clinic, the most common being flaccid and spastic. Speech symptoms, including velopharyngeal dysfunction, depend on the particular combinations of dysarthria in the individual patient.

Neurogenic causes: Developmental verbal dyspraxia (DVD)

• Terminology Developmental verbal dyspraxia (DVD) There is no specific definition or agreed set of diagnostic characteristics for DVD but there is consensus about the types of features often seen, indicating a deficit in the planning and programming of movements for speech. For more detailed discussion see RCSLT Policy Statement, Developmental Verbal Dyspraxia, 2011.

Velopharyngeal dysfunction can be a feature of dyspraxic speech and symptoms may include disturbances of nasal resonance and airflow. Just as there are likely to be difficulties in the neuromotor planning and programming for articulation, it follows that the velopharyngeal mechanism can be affected (Weiss et al., 1987).

Mislearning This refers to incorrect opening and closing of the velopharyngeal port in the absence of a structural or neurological problem. Peterson (1975) describes nasal emission as a component of the misarticulation of fricatives and affricates, both in children who had been treated for cleft palate and in children with no history of palatal structural problems. Either a previous history of true physiological velopharyngeal dysfunction or fluctuating hearing loss could

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easily be a basis for ‘mislearning’ how to produce normal fricative and affricate sounds. However, it is not clear what may explain its occurrence in children without such histories. Any or all fricatives and affricates may be misarticulated as active nasal fricatives. Typically the /s/ and /z/ consonants are most frequently affected as well as /f v ʃ ʧ ʤ z/. Affricates, which are generally acquired later in developmental terms, are sometimes correctly articulated when /s/, acquired earlier, is not. Finally, for individuals with hearing impairment, a significant degree of hearing loss results in an inability to establish nasal versus non-nasal contrasts in speech, leading to a habitual pattern of abnormal resonance.

What speech features are associated with VPD? Resonance Hypernasal resonance, or excessive nasal resonance in speech (Grunwell & Sell, 2001), is a significant perceptual feature of VPD, most noticeable on vowels and approximants. Vocal resonance can be described as the amplification of sound waves produced by the vibrations of the vocal cords (Witzel, 1995). It manifests as a perceptual quality relating to the balance of air in the oral and nasal cavities during speech (Britton, 2004). In severe hypernasality, voiced plosives /b, d, g/ in English may be sufficiently weakened that they are perceived as the nasals /m, n, ŋ / (Sell et al., 1994).

Nasal airflow In addition to abnormal resonance, VPD may also lead to abnormal airflow. Where VP closure is incomplete, air passes through the nasal cavity and is perceived as nasal emission and/or nasal turbulence, accompanying oral consonants (D’Antonio & Scherer, 1995). Airflow may be entirely nasal or simultaneously oral and nasal, depending on the extent of velopharyngeal opening and the forcefulness of airflow. Nasal emission can be audible, with frication heard as the air flows through the nasal cavity, or may be inaudible and only detected clinically by observing fogging or misting of a small mirror held under the nostrils during speech (Peterson-Falzone et al., 2006). Where the velopharyngeal gap is relatively small, a distinctive turbulent noise is perceived (Kummer et al., 1992). This often accompanies plosive and

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fricative targets (Sell et al., 1994) and can be highly distracting to listeners (McWilliams et al., 1990). This feature is now generally referred to as nasal turbulence, but has also been described as nasal snort, nasal rustle and nasal friction (Grunwell & Sell, 2001).

Nasal/facial grimace This is believed to be a compensatory behaviour by the speaker that occurs subconsciously. The speaker constricts their nostrils and sometimes other facial muscles in an attempt to inhibit abnormal airflow through the nose (Grunwell & Sell, 2001). Grimace may therefore serve as an indicator of anatomical limitations or be part of velopharyngeal mislearning. There are differing degrees of grimace: nasal flare, nasal grimace involving the nostrils and the upper lip, and facial grimace which includes the mid and upper face.

Speech sound production VPD results in insufficient intraoral pressure to produce oral consonants (Theodorus & Thompson-Ward, 1998). Sounds are produced as their nasal equivalent, e.g., as in [mɔl] for ‘ball’ and [nɒl] for ‘doll’ or therefore as weak oral productions, [b ͉ɔl] and [d ͉ɒl]. Compensatory articulations may also be adopted by the speaker in the presence of VPD. These are non-English realisations of English consonants (Wyatt et al., 1996). They are learned articulation deviations, adopted in the course of speech acquisition and are believed to result from strategies developed by the child to ‘functionally offset the structural impediments imposed by VPD’ (Peterson-Falzone et al., 2006). The following consonants (compensatory articulations) are produced non-orally and can therefore be produced in the presence of VPD.

Glottal plosives A glottal plosive is produced by abrupt adduction and release of the vocal folds. It is usually produced as a substitution for plosive consonants. Videofluoroscopy shows that there is little or no velopharyngeal movement when a glottal plosive is made.

Pharyngeal plosives and fricatives Pharyngeal plosives are produced by the dorsum of the tongue moving back

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

to contact the pharynx, causing pressure to build up with a sudden release of air. Pharyngeal fricatives are made when the dorsum of the tongue moves backwards towards the pharynx to constrict the airstream, causing friction.

Nasal fricatives Fricative and/or affricate consonants are sometimes replaced by a voiceless nasal consonant (all airstream is directed nasally).

What other problems are associated with VPD? Nasal regurgitation/reflux Individuals with VPD may experience regurgitation of liquid and food into the nose (Marsh, 2009). This most frequently occurs with foods such as chocolate and yoghurt and similar textures. Recurrent and chronic sinus infections

Figure 2.2 Articulation and airflow in nasal fricative

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can also be a sign of nasal reflux, resulting from repeated escape of food and drink into the nasal cavity. Persistent middle ear effusion may also be related to nasal reflux, extending up the Eustachian tube and through the middle ear (Devani et al., 1999).

Voice disorders In addition to difficulties with consonant production, it has been suggested that speakers with VPD are prone to voice disorders (Bzoch, 1989 in Wyatt et al., 1996). Leder and Lerman (1985) and Warren (1986) suggest that the speaker learns to use increased laryngeal adductory and respiratory forces to compensate for air escape due to VPD. Other authors have hypothesised that voice difficulties arise from hyperfunction associated with speech articulations such as pharyngeal and glottal stops, laryngeal fricatives and affricates and posterior tongue posturing (Tanimoto et al., 1994; Kido et al., 1992). Establishing the relationship between VPD and dysphonia is difficult as there are likely to be multiple aetiological factors, as with the general dysphonic population. More detailed reference to specific voice disorders, assessment and treatment techniques can be found in Chapter 6. g Further Reading: Aronson and Bless (2009); Sherwell (2009); Hartnick and Boseley (2008); Hunt and Slater (2003); Colton, Casper and Leonard (2006); Andrews and Summers (2002); Mathieson (2001).

Stress VPD This is an infrequent problem, but every so often brings a challenge to UK Cleft Centres. Weber and Chase (1970) describe an oboe player who had nasal air loss after ten minutes of continuous play. The authors labelled this as ‘stress velopharyngeal incompetence’ where the demand placed on the VP mechanism exceeds its capacity. It is estimated that intraoral pressures to play a muted trombone are around thirty times greater than that required for speech (Dibbell et al., 1979). The implications of stress VPD for the professional musician are therefore significant and require careful evaluation and management. g Further Reading: Evans et al. (2009). The role of the soft palate in woodwind and brass playing. International Symposium on Performance Science.

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Impact of VPD The primary impact of VPD (cleft and non-cleft) is on speech, although nasal regurgitation of food and drink and nasal or facial grimacing can also be features, as previously discussed. A hypernasal speech quality caused by VPD sounds noticeably abnormal to the listener, making speech weak, indistinct and difficult to understand. In addition, the development of oral pressure consonants may be restricted and compensatory articulation can make speech abnormal. It is accepted that hypernasal speech often results in a negative impact on quality of life for children and adults (Barr et al., 2007; Deary et al., 2003; Lallh & Rochet, 2000). Quality of Life (QoL), is defined by the World Health Organization as an: “Individual’s perception of their position in life in the context of the culture and value systems in which they live and in relation to their goals, expectations, standards and concerns” Group, W.H.O.Q.O.L., 1993, p.153. Health Related Quality of Life (HRQOL) is generally considered to be part of the broader construct of Quality of Life and is defined as the impact that a particular health condition (or intervention) has on an individual (Petersen, 2003). Items related to objective (health status) aspects of HRQOL tend to focus on physical symptoms, functioning and activity limitations, whereas items related to subjective (well-being) aspects of HRQOL focus on feelings, concerns and worries about the condition (Eiser & Morse, 2001).

What is the evidence?

• Research

Barr et al. (2007) devised the Velopharyngeal Insufficiency Quality of Life measure (VPIQOL). Their objective was to determine whether there are alterations in QOL in children with VPI aged 5–17 years, and if there was agreement between children and their parents in perceived quality of life. They compared a generic QoL tool (Pediatric Quality of Life Inventory, version 4) with the VPIQOL using a control group for all domains on

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both measures. They found that children with VPI and their parents both reported more negatively affected QoL. Key findings were: Speech limitations were the most affected domain in the VPIQOL for both parents and children in the VPI group. Children with VPI and their parents ranked ‘speech as nasal’, ‘difficulty being understood’ and ‘speech sounds abnormal' as high, with ‘speech is difficult for strangers to understand’ as being the highest affected. In addition, parents of children with VPI included ‘difficulty being understood at school’, ‘difficulty being understood over the phone’, ‘gets frustrated and/or gives up when he/she is not understood’ and ‘I am concerned about my child’s nasal speech’. Damiano et al. (2007) also showed that speech and aesthetic concerns seem to be important factors affecting health-related quality of life for children with oral clefts. These factors were shown to be more important to children as they get closer to adolescence, i.e., aged 8–12 years. Similarly, a study by Hunt et al. (2006) showed that children and adults with a cleft, aged 8–21, were less happy with their speech than those in control groups. Broder et al. (1992) found some indication that satisfaction with speech increases with age and Richman (1983) also reports age-related differences and concerns when it comes to the individual’s satisfaction with speech. More recently, Berger and Dalton (2011) reported that perceived speech problems are an important factor relating to children’s adjustment: “Good adjustment was associated with more positive social experiences, better maternal adjustment, greater satisfaction with appearance, fewer perceived problems with their speech, lesser avoidant coping strategies (social withdrawal and distraction) and being female.” p.88 Family relationships, peer relationships, peer rejection, teasing, bullying, play, school functioning and integration in school are all recurrent themes in the Markham et al. (2009) study of children with speech, language and communication needs and their perceptions of their quality of life. These areas relate to the concepts of activity and participation and are central in considering quality of life. They form part of the International Classification of Functioning, Disability and Health (ICF) framework proposed by the World

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Table 2.2 Activity and participation in relation to speech, language and communication skills Activity

Participation

Being intelligible (clarity of speech)

Being able to take part in family life/events

The ability to hold a conversation

The ability to form relationships with peers

The ability to express needs and wants

The ability to interact with the community

Being able to read and spell

Being able to achieve goals in life

Health Organization (WHO, 2001), where activity and participation feature alongside body structures and functions in an attempt to focus on impact and not simply disorder. McLeod (2004) has reported preliminary work in applying these concepts to speech impairment, using the definitions shown in Table 2.2. Whether speech impairment is lasting or transient, the impact it has on a person’s self-image or will to communicate is unpredictable. It is acknowledged that the same type and degree of speech impairment can have differing effects on individuals, yet different types or degrees of impairment can result in almost the same impact on an individual’s participation in society. What is apparent is that there are other factors that influence how much people’s lives will be influenced by their speech impairment. As Havstam and Lohmander (2011) state, the implication is that any assessment of VPD should consider the impact on the individual and their ‘communicative participation’. They also point out that the effect of hypernasal speech on the individual may not necessarily correlate with its severity; in some individuals a relatively mild nasal voice quality or articulatory deviation can lead to low self-esteem and lack of participation in educational and social situations.

References Barr, L., Thibeault, S., Harlan Muntz, M. & de Serres, L. (2007). Quality of life in children with velopharyngeal insufficiency. Archives of Otolaryngology – Head & Neck Surgery, 133, 224–229. Berger, Z.E. & Dalton, L.J. (2011). Coping with a cleft II: Factors associated with psychosocial adjustment of adolescents with a cleft lip and palate and their parents. The Cleft PalateCraniofacial Journal, 48(1), 82–90. Britton, L. (2004). Speech and language difficulties associated with cleft palate. In V. Martin

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& P. Bannister (Eds), Cleft Care: A Practical Guide for Health Professionals on Cleft Lip and/ or Palate (pp.95–103). Wiltshire: APS. Broder, H.L., Smith, F.B. & Strauss, R.P. (1992). Habilitation of patients with clefts: Parent and child ratings of satisfaction with appearance and speech. The Cleft Palate-Craniofacial Journal, 29(3), 262–267. Bzoch, K. (1989). Communicative Disorders Related to Cleft Lip and Palate, 3rd Edn. Boston: Little, Brown. In R. Wyatt, D. Sell, J. Russell, A. Harding, K. Harland & E. Albery (1996), Cleft palate speech dissected: A review of current knowledge and analysis. British Journal of Plastic Surgery, 49, 143–149. Croft, C., Shprintzen, R. & Ruben, R. (1981). Hypernasal speech following adenotonsillectomy. Archives of Otolaryngolology­– Head and Neck Surgery, 89(2), 179–188. Damiano, P., Tyler, M., Romitti, P., Momany, E., Jones M., Canady, J. & Murray, J. (2007). Health Related Quality of Life among preadolescent children with oral clefts: The mother’s perspective. Pediatrics, 120(2), 283–290. D’Antonio, L. & Scherer, N. (1995). The evaluation of speech disorders associated with clefting. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach (pp.176–220). St Louis, MO: Elsevier Mosby. Darley, F., Aronson, A. & Brown, J. (1969a). Clusters of deviant diagnostic patterns of dysarthria. Journal of Speech and Hearing Research, 12, 462–496. Darley, F., Aronson, A. & Brown, J. (1969b). Differential diagnostic patterns of dysarthria. Journal of Speech and Hearing Research 12, 246–269. Darley, F., Aronson, A. & Brown, J. (1975). Motor Speech Disorders. Philadelphia: WB Saunders. Deary, I., Wilson, J., Carding, P. & Mackensie, K. (2003). The dysphonic voice heard by me, you and it: Differential associations with personality and psychological distress. Clinical Otolaryngology & Allied Sciences, 28, 374–378. Devani, P., Watts, R. & Markus, A.F. (1999). Speech outcome in children with cleft palate: Aerophonoscope assessment of nasal emission. Journal of Cranio-Maxillofacial Surgery, 27(3), 180–186. Dibbell, D., Ewanowski, S. & Carter, W. (1979). Successful correction of velopharyngeal stress incompetence in musicians playing wind instruments. Plastic and Reconstructive Surgery, 64, 662–664. Duffy, J. (1995). Motor Speech Disorders: Substrates, Differential Diagnosis, and Management, 2nd Edn. St Louis: Elsevier Mosby. Dworkin, J.P. & Johns Donnell, F. (1980). Management of velopharyngeal incompetence in dysarthria: A historical review. Clinical Otolaryngology & Allied Sciences, 5(1), 61–74. Eiser, C. & Morse, R. (2001). Can parents rate their child’s health related quality of life? Results of a systematic review. Quality of Life Research, 10, 347–357. Group, W.H.O.Q.O.L. (1993). Study protocol for the World Health Organization project to develop a Quality of Life assessment instrument (WHOQOL). Quality of Life Research, 2, 153­–159.

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Grunwell, P. & Sell, D. (2001). Speech and cleft palate/velopharyngeal anomalies. In A. Watson, D. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Havstam, C. & Lohmander, A. (2011). Communicative participation. In S. Howard & A. Lohmander (Eds), Cleft Palate Speech Assessment and Intervention (pp.305–312). Chichester: Wiley-Blackwell. Helmi, C. & Pruzansky, S. (1980). Craniofacial and extracranial malformations in the Klippel-Feil syndrome. Cleft Palate Journal, 17(1), 65–88. Henningson, G. & Isberg, A. (1988). Influence of tonsils on velopharyngeal movements in children with craniofacial anomalies and hypernasality. American Journal of Orthodontics and Dentofacial Orthopedics, 94, 253–261. Hunt, O., Burden, D., Hepper, P., Stevenson, M. & Johnson, C. (2007). Parent reports of psychosocial functioning among children and adults with cleft lip and palate. Cleft Palate Craniofacial Journal, 43, 598­–605. Isberg, A. & Henningson, G. (1985). Influence of palatal fistulae on velopharyngeal movements: A cineradiographic study. Plastic and Reconstructive Surgery, 79, 535–530. Johns, D., Rohrich, R. & Awada, M. (2003). Velopharyngeal incompetence: A guide for clinical evaluation. Plastic and Reconstructive Surgery, 112, 1890–1898. Kido, N., Kawano, M. & Tanokuchi, F. (1992). Glottal stop in cleft palate. Studia Phonologica, 26, 34–41. Kummer, A., Curtis, C., Wiggs, M., Lee, L. & Strife, J. (1992). Comparison of velopharyngeal gap size in patients with hypernasality, hyponasality and nasal emission and, or nasal turbulence (rustle) as the primary speech characteristic. Cleft Palate-Craniofacial Journal, 29, 152–156. Lallh, A. & Rochet, A. (2000). The effect of information on listeners’ attitudes toward speakers with voice or resonance disorders. Journal of Speech Language and Hearing Research, 43, 782–795. Leder, S. & Lerman, J. (1985). Some acoustic evidence for vocal abuse in adult speakers with repaired cleft palate. The Laryngoscope, 95, 837–840. McLeod, S. (2004). Speech pathologists’ application of the ICF to children with speech impairment. International Journal of Speech-Language Pathology, 6, 75–81. McWilliams, B., Morris, H. & Shelton, R. (1990). Cleft Palate Speech, 2nd Edn. Philadelphia, Toronto: BC Decker. Markham, C., Van Laar, D., Gibbard, D. & Dean, T. (2009). Children with speech, language and communication needs: Their perceptions of their quality of life. International Journal of Language & Communication Disorders, 44(5), 748–768. Marsh, J.L. (2009). Velo-pharyngeal dysfunction: Evaluation and management. Indian Journal of Plastic Surgery: Official Publication of the Association of Plastic Surgeons of India, 42 (Suppl), S129. Mason, R., Turvey, T. & Warren, D. (1980). Speech considerations with maxilliary advancement procedures. Journal of Oral Surgery, 38, 752–758.

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Mathieson, L. (2001). Greene and Mathieson’s The Voice and Its Disorders, 6th Edn. London: Whurr Publishers. Morris, H.L., Wroblewski, S.K., Brown, C.K. & Van Demark, D.R. (1990). Velarpharyngeal status in cleft palate patients with expected adenoidal involution. The Annals of Otology, Rhinology, and Laryngology, 99 (6 Pt 1), 432. Petersen, P. (2003). The World Oral Health Report 2003. Continuous improvement of oral health in the 21st century – the approach of the WHO Global Oral Health Programme. World Health Organization, 2003. Available at: http://www.who.int/oral_health/media/ en/orh_report03_en.pdf. Accessed December 31, 2012. Peterson, S. (1975). Nasal emission as a component of the misarticulation of sibilants and affricates. Journal of Speech and Hearing Disorders, 40, 106–114. Peterson-Falzone, S., Trost-Cardamone, J., Karnell, M. & Hardin-Jones, M. (2006). The Clinician’s Guide to Treating Cleft Palate Speech. St. Louis: Elsevier Mosby. RCSLT. (2011). Developmental Verbal Dyspraxia: Policy Statement, RCSLT London. Richman, L.C. (1983). Self-reported social, speech, and facial concerns and personality adjustment of adolescents with cleft lip and palate. Cleft Palate Journal, 20(2), 108–112. Saunders, N., Hartley, B., Sell, D. & Sommerlad, B. (2004). Velopharyngeal insufficiency following adenoidectomy. Clinical Otolaryngology Allied Science, 29, 686–688. Sell, D., Harding, A. & Grunwell, P. (1994). A screening assessment of cleft palate speech: “GOS.SP.ASS” (Great Ormond Street Speech Assessment). European Journal of Disorders of Communication, 29, 1–15. Shprintzen, R., Sher, A. & Croft, C. (1987). Hypernasal speech caused by tonsillar hypertrophy. International Journal of Pediatric Otolaryngology, 14, 45–56. Tanimoto, K., Henningsson, G., Isberg, A. & Ren, Y.F. (1994). Comparison of tongue position during speech before and after pharyngeal flap surgery in hypernasal speakers. Cleft Palate-Craniofacial Journal, 31, 280–286. Theodorus, D. & Thompson-Ward, E. (1998). Treatment of dysarthria. In B.E. Murdoch (Ed.), Dysarthria. Cheltenham: Stanley Thornes Ltd. Trost-Cardamone, J. (1989). Coming to terms with VPI: A response to Loney and Bloem. Cleft Palate Journal, 26, 68–70. Van Demark, D.R., Hardin, M.A. & Morris, H.L. (1988). Assessment of velopharyngeal competence: A long-term process. Cleft Palate Journal, 25(4), 362–373. Walter, V., Nisa, L. & Leuchter, I. (2012). Acute isolated velopharyngeal insufficiency in children: Case report and systematic review of the literature. European Archives of OtoRhino-Laryngology, 269, Online publication 09/10/2012. http://link.springer.com.eresources. shef.ac.uk/search?query=VELOPHARYNGEAL&facet-publication-title=%22European+A rchives+of+Oto-Rhino-Laryngology%22&search-within=Journal Warren, D. (1986). Compensatory speech behaviours in individuals with cleft palate: A regulation/control phenomenon? Cleft Palate Journal, 23, 251–260. Weber, J. & Chase, R. (1970). Stress velopharyngeal incompetence in an oboe player. Cleft Palate Journal, 7, 858–861.

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Weiss, C., Gordon, M. & Lillywhite, H. (1987). Clinical Management of Articulatory and Phonologic Disorders. Baltimore: Williams and Wilkins. Witt, P.D., Myckatyn, T., Marsh, J.L., Grames, L.M. & Dowton, S.B. (1997). Need for velopharyngeal management following palatoplasty: An outcome analysis of syndromic and nonsyndromic patients with Robin sequence. Plastic and Reconstructive Surgery, 99, 1522–1534. Witt, P.D., Myckatyn,T. & Marsh, J.L. (1998). Salvaging the failed pharyngoplasty: Intervention outcome. The Cleft Palate-Craniofacial Journal, 35(5), 447–453. Witzel, M., Rich, R., Margar-Bacal, F. & Cox, C. (1986). Velopharyngeal insufficiency after adenoidectomy: An 8 year review. International Journal of Pediatric Otorhinolaryngology, 11, 15–20. Witzel, M. (1995). Communicative impairment associated with clefting. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach, (pp.37–166). Boston: Elsevier Mosby. World Health Organization (WHO). (2001). ICF: International Classification of Functioning, Disability and Health. Geneva, Switzerland: WHO. Wyatt, R., Sell, D., Russell, J., Harding, A., Harland, K. & Albery, E. (1996). Cleft palate speech dissected: A review of current knowledge and analysis. British Journal of Plastic Surgery, 49, 143–149.

Bibliography Andrews, M. & Summers, A. (2002). Voice Treatment for Children and Adolescents. San Diego CA: Singular Thomson Learning. Aronson, A. & Bless, D. (2009). Clinical Voice Disorders, 4th Edn. New York: Thieme. Colton, R., Casper, J.K. & Leonard, R. (2011). Understanding Voice Problems: A Physiological Perspective for Diagnosis and Treatment, 4th Edn. Philadelphia, PA: Lippincott Williams & Wilkins. Evans, A., Ackermann, B. & Driscoll, T. (2009). The role of the soft palate in woodwind and brass playing. International Symposium on Performance Science. Utrecht, The Netherlands: European Association of Conservatoires, pp.267–272. Hartnick, C. & Boseley, M. (2008). Paediatric Voice Disorders. San Diego, CA: Plural Publishing. Howard, S. & Lohmander, A. (2011). Cleft Palate Speech Assessment and Intervention. Chichester: Wiley-Blackwell. Hunt, J. & Slater, A. (2003). Working with Children’s Voice Disorders. London: Speechmark. Sherwell, C. (2009). Voice Work: Art and Science in Changing Voices. Chichester: John Wiley and Sons Ltd.

3 Section 1 Oral Examination Fiona Jeyes

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Aim of this section This section focuses on oral examination and the reasons why this is so critical in the assessment of children with speech disorders. The procedure for carrying out the examination is explained with a particular focus on the identification of submucous cleft palate.

Why should speech and language therapists look in children’s mouths? Where an individual presents with a speech problem, an orofacial examination should be routinely carried out. If children are difficult to understand, an examination of the speech mechanism should always be undertaken, to ascertain that anatomical factors are not present which may be detrimental to speech development. Stenglehofen (1999) p.26. The aim of an oral assessment is therefore to exclude any structural anomaly which would prevent a child from easily building a complete sound system. However, it may be that oral examination is no longer an automatic part of initial assessment for a generation of UK therapists educated in the importance of phonological theory to explain children’s sound system errors and substitutions. Every year children are referred to cleft palate centres with a restricted sound system and found to have a submucous cleft palate. More worrying is that a child may be referred to the cleft palate centre with a previously undiagnosed, frank cleft palate. Often these children have previously seen a range of health professionals who have not discovered their structural problem. The oral examination takes a few minutes and can usually be forgotten once the results are recorded. Most children will show you their mouth without worry; the more confident the therapist is, the better. As a therapist you become confident by repeating procedures frequently. Only by looking in lots of mouths that work normally is it possible to be sure about things that are unexpected or out of the range. A basic oral examination will be visual, with

Oral Examination

29

nothing placed in the child’s mouth. Only if you suspect there is a problem with the palate would you need to continue to something more invasive. Oral examination should also be carried out for children who have a repaired cleft palate. This will give information about how easily the soft palate is moving and whether any fistula remains in the hard palate or alveolus. A speech and language therapist may be seeing a child whose cleft repair is incomplete and it is important to know what constraints the oral structure is placing on that child. Information from oral examination should be integrated with the rest of your assessment of the child. It is possible for a child to have a submucous cleft palate, but to have no issues with making plosives and fricatives, and have normal resonance with no escape of air from the nose. In this case no action would need to be taken except to inform the parents and GP, so that the correct referral could be made if the situation changes for the child (for example, when the adenoids shrink, or when the pubertal growth spurt occurs). Oral examination will also give you a feel for how the child is able to modify movement of the articulators to request and how smoothly they can make those movements. You can see how well their teeth slot together and how comfortable they are with making a lip seal and maintaining it. This will give you valuable information about some of the background to their current articulatory competence. You should also find out about feeding history (including information about infant milk feeding), and current eating and drinking at the same time.  Resource CD: Results could be noted on a checklist: see CD resources.

• Equipment for oral examination (Figure 3.1.1)

This is not a sterile procedure, but it is important that your hands and equipment are clean before and after the examination. 1. Light. There are various types of pen torch available, both disposable and reusable. They are easy to carry and to wipe off after use. It may help to have two torches so that the child can play with one before you need to use it. There are also novelty torches from toy manufacturers which provide a bright beam and are attractive to children. If you can position the child to take advantage of the natural light in the room that is a good idea too.

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Depending what you see from your visual check you may also need: 1. Gloves. Essential if you are going to put your fingers in the child’s mouth. 2. Tongue depressors. Plain wooden tongue depressors can be bought cheaply and carried in a zipped wallet so they are clean. There are also individually wrapped and flavoured ones which can be bought more expensively on line. Any of them feel nicer on the tongue if placed in some cold water before use. 3. A reflective surface to check for nasal escape of air (e.g., a small dental mirror or plastic detail reflector).

Procedure Before starting the oral examination you will have to decide whether the child would be more comfortable seated on a parent’s lap, or on their own

Figure 3.1.1 Oral examination kit

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chair. Sometimes leaving them where they are makes the situation seem more routine. Parents are sometimes surprised at how readily children participate in this and the child may become aware of their unease and react to it. The more commonplace you can make it the better your chance of having a good look. You can get lots of information about the child by observing him before you get to the oral examination: • Consider whether the face is symmetrical and expressive. • If he is a mouth breather are there signs of nasal blockage? (Look for blue shadows under the eyes; bridge of nose disappearing due to puffy skin alongside; hyponasal voice tone.) • Is saliva controlled during playing and talking? • You may also become aware if the child is able to make movements naturally, but then struggles when trying to do them to your request.

Lips 1. Can he hold his lips shut? 2. Is his smile symmetrical? 3. Can he purse his lips? Having looked at these things you should then consider the reasons for inability to do any of them. A child who has a repaired cleft lip may have an upper lip that is short or tight and is not able to meet with the lower lip. A child who has blocked nasal airways may be reluctant to close his lips, as he will not be able to breathe. A child with poor muscle tone may not be able to make their lip muscles hold together. They might try to compensate for this by using their teeth to hold the lower lip, or make a very loose closure of the lips. It is also possible for the underlying structures to affect the position of the lips and their ability to close. The information you have might also help in planning which sounds to target in the first instance.

Tongue 1. Can he protrude his tongue outside his mouth without the support

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

of his lower lip? Does the tongue sit centrally outside his mouth? Is it fairly still or does it move lots? If moving, what sort of movement do you see? 2. Can he move his tongue easily from one corner to the other of the mouth? 3. Can he turn the tip up towards his top lip/nose? Once again you get an idea of whether movements are symmetrical and how much effort is needed for them. If you find the child has a tongue tie you need to think whether this is significant.

• Note Tongue tie There is little good research on the effect on speech caused by ‘ankyloglossia’ (Kummer, 2005). Babies are frequently treated at birth to aid feeding as breastfeeding can be adversely affected (Messner et al., 2000). Overall, the literature suggests that most people do not have poor speech consequences from a tongue tie. However, if there are other oro-motor difficulties, or if the child has specific difficulty with tongue tip sounds ‘frenulectomy’ might be considered (Messner & Lalakea, 2002). It is important that parents are aware of which sounds might be improved by the operation and do not think that loosening the tie will correct major speech sound or language difficulties.

Teeth 1. Primary/mixed/secondary dentition? 2. How do teeth bite together? Normal bite, anterior open bite, Class III malocclusion? 3. What condition are the teeth in? If teeth have been removed how long ago was this? 4. What shape are the arches?

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• Terminology An anterior open bite is defined by a vertical space between the upper and lower front teeth. A Class II malocclusion is described when the top jaw and teeth severely overlap the lower jaw and teeth, leaving a significant gap. A Class III malocclusion is when the top jaw and teeth close behind the lower ones so that the lower teeth overlap the top ones and the lower jaw protrudes.

Teeth are generally not a major factor in children’s ability to make sounds, but a child who has had no incisors since he was two years of age will have had a different experience of tongue movements than a child whose teeth form a barrier for the tongue to touch. Children who have had a cleft palate sometimes have very narrow dental arches which can affect tongue placements. If teeth are rotated or erupting in the wrong place they may have an impact on where the tongue goes to make sounds.

Hard palate 1. Is it intact? If you see a fistula, describe where it is and what shape. Does it go through to the nose? The family and child may know if air can be pushed through the hole, or if food gets through to the nose. A fistula in the alveolar region may mean that a child retracts the tongue in the mouth to make sounds more efficiently. This may lead to the substitution of palatal or velar sounds. 2. What shape is the palate? If very narrow and high this may suggest some difficulty with tongue movement or position as the tongue exerts a shaping force on the hard palate. Surface anatomy of the hard palate is rarely significant and seams and bumps are common. Some people have dimples near the join to the soft palate.

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Figure 3.1.2 Central split evident in palate, muscles running up towards the hard palate, rather than towards the midline

Soft palate 1. When the child says /a:/ what happens to their soft palate? Does the palate move up briskly and equally on both sides? Is there any movement? Do you notice if the pharyngeal walls move too? 2. Is the palate all pink and healthy–looking? 3. What does the uvula look like? A normal soft palate will form a dome shape on phonation. You do not really see where the muscles are; everything is pink, opaque and firm. Many people have an obvious single white seam line going down the middle of their palate, but this does not indicate a problem. While looking at the soft palate you will probably get a view of the tonsils. In some children they are large enough to constrain the soft palate from easy movement. In others they may promote a forward tongue posture to enable as much airway as possible. Look out for a blue, thin, translucent area in the middle of the palate (zona pellucida). This suggests that the muscle band is not meeting in the centre of the palate. Sometimes you see the muscles going from the end of the soft palate towards the hard palate in a ‘^’ shape.

Oral Examination

Figure 3.1.3 Bifid uvula, zona pellucida, enlarged tonsils

Figure 3.1.4 Bifid uvula, transparent skin in the centre, slight ‘^’ impression

Figure 3.1.5 Still counts as bifid uvula

35

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

It can be difficult to get a good look at the uvula so it is worth putting in some preparation before you try. 1. Position the child well so you have the light. 2. Ask the child to practise saying a good loud /a:/ sound, then get them to look up at the ceiling, open wide and say their /a:/ for you. You may need to use a tongue depressor to keep the back of the tongue down and out of the way. 3. Is it a single uvula or split in two? If there is a single uvula can you see to the end? 4. Sometimes the signal of a submucous cleft is a groove on the uvula, rather than an actual split (see Figures 3.1.3 to 3.1.5).

• Note

Incidence of bifid uvula/SMCP in population Bifid uvula is surprisingly common and often asymptomatic. An early study of 10,836 schoolchildren in Denver (Stewart et al., 1972) found 100 bifid uvulas and nine submucous cleft palates (confirmed on re-examination by a cleft palate surgeon). Other studies quote levels between 1%–2% for Caucasians. If you have seen any of these features now is the time to get the gloves on! Feel along the central line of the hard palate to where it becomes the soft palate. Children are less likely to gag if you press quite firmly and move back smoothly. You need to go back to about level with the last tooth. Is the edge between the hard and soft palate smooth or can you feel a notch?

Signs of a submucous cleft palate • Bifid uvula • Zona pellucida • Notch in hard palate • Incorrectly positioned muscles.

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37

A submucous cleft palate (SMCP) is one where the surface is complete, but the underlying structures are not correctly aligned and joined. For many children it will cause the same speech symptoms as having a cleft palate. There will often be a history of difficult infant feeding; some children struggle to breastfeed but manage with a bottle. Weaning often comes as a relief to these babies and their parents. Speech will show the results of having a velopharyngeal mechanism that is not able to make adequate closure, either in the production of weak pressure sounds or in compensatory sounds, which can make speech hard to understand. For example, some children will use glottal stops in place of consonants in all word positions. However, these children will not sound nasal as they are not attempting an oral sound. Some children are able to develop normal speech and language in the presence of an obvious SMCP, while others struggle with a structure that appears ‘nearly right’. It is vital that speech and language therapists make sure that there is no structural deficit before they embark on sound work that cannot be successful.

• Case Study: Joel

Joel was first seen by a speech and language therapist aged 5 years 4 months. He had been referred by his school staff who were concerned about his extremely poor clarity. Despite his speech difficulties he was doing well with developing reading. He had a history of ear infections and had been seen frequently by his GP. His mother had been concerned about his speech for years but had been given different reasons by health professionals about why he was not developing clarity. No health worker had made a referral to speech therapy. The speech and language therapist noted that Joel was not able to hear her during their session and was using lip reading to cope in the assessment. Conversational speech was unintelligible to the therapist although mum was able to understand most of what he said.

Assessment results • Speech showed intact syllable structure

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Figure 3.1.6 Joel is now 10 and has clear speech following surgical repair of his cleft palate and insertion of grommets. He has had SLT programmes, which worked on introducing groups of sounds, starting with plosives and moving to fricatives. He had particular difficulty achieving velar placement and needed use of a ‘novel’ sound approach for these sounds (see Chapter 7).

• Consonants /m/ and /n/ were used (no velar nasal) • There were no oral plosives or fricatives present in any word position • Glottal stops were prevalent in word final position • The therapist thought that some of the time Joel sounded hyponasal • There was no nasal emission of air, or nasal resonance. The therapist went on to an oral examination. There was nil significant in lips, tongue or teeth. Joel’s hard palate appeared mostly intact, but opened out into a wide cleft of the soft palate. Joel’s therapist might have felt that an oral check was not necessary as his doctor had seen him on many occasions. His compensatory articulation pattern meant that he did not sound nasal and did not have air coming from his nose as he spoke. Using a mirror under his nose would not have shown any misting as he was not using oral consonants and therefore did not have nasal escape. Happily for Joel, his therapist checked out his oral structures on the

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first occasion that she met him and discovered his cleft palate. She then referred him urgently to the local cleft palate service.

• Case study: Ned

Ned was referred to the cleft palate team aged 9. Ned’s family had moved around and he had lived in several countries. His medical history was complex and his records had not followed him. Ned had a stormy early history after a premature birth. He had great difficulty with milk feeding and had nasal regurgitation of fluids. Solid feeding developed well. He was given grommets when he was 2, but hearing had remained a problem for him. His language development was delayed and he needed SLT intervention over a long period. Between ages 5 and 7 he received intensive speech therapy for disordered speech. A diagnosis of dyspraxia had been suggested. Ned had a diagnosis of ADHD (Attention Deficit Hyperactivity Disorder) and was prescribed medication for this. He was also treated for asthma. Ned reached the cleft palate service after his paediatrician referred him to ENT (Ear, Nose & Throat) locally. The ENT surgeon referred him to the centre after looking into his mouth. The speech therapy assessment was done in the cleft centre.

Assessment results: • Ned was articulating all sounds correctly, except /l → j/ • Hypernasal resonance on all vowels. Consonants were weak • Nasal turbulence and nasal emission accompanied a range of sounds • Mirror testing showed emission through both nostrils on syllables containing close and open vowels (usually tested using repetitions of /pa, pi, ka, ki, s:/) • All plosives and fricatives were improved when Ned said them with his nostrils blocked.

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Oral examination showed: • A bifid uvula • A blue, thin middle section of the soft palate • Apparent incorrect muscle alignment • A notch in the hard palate • A palate that appeared short (or a deep pharynx). These features gave a firm diagnosis of submucous cleft palate. In fact, Ned showed all the features looked for in the assessment. Six months after surgery to repair his palate Ned achieved normal resonance and clarity. His mother reports that he is more communicative and confident in school. Ned had a history of speech and language disorder going back to infancy. Therapists had worked hard with him and his family to address his difficulties. This type of speech disorder means it is often hard to recognise when problems are structural or developmental. However, an oral examination as part of routine assessment before starting any sound work might have prevented many years of speech frustration for Ned and his family.

• Case study: Maia

Maia was referred to the cleft service by her local SLT aged 2½ years. Her initial referral was for language delay. She was a good communicator, using all the channels available to her. Maia’s hearing status was not confirmed. Her mother felt that she misheard like-sounding words (e.g., bite/bike).

Assessment results Her therapist had noted no oral consonants. Maia used nasalised vowels and had audible nasal emission when blowing. Maia had a heart-shaped tongue and low-set round ears. The therapist had been unable to view Maia’s palate. At initial assessment with the cleft palate centre therapist the following were recorded:

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• Maia had not been able to breastfeed, but had managed a bottle. She had frequent occurrences of milk escaping from her nose when feeding. Weaning went well. All developmental milestones were delayed. • Maia was a quiet baby, little babble, only started to chatter around 2 years of age. • Maia used only /m, n/ nasalised vowels and an attempt at a fricative word finally. • Good vowel differentiation, intonation and stress. Quite easy to understand, despite the lack of consonants. • Oral exam showed reduced tone of all facial muscles, including lips. • Anterior open bite despite restricted dummy use. Lips not really closing tightly. Some tongue movement seen. • Uvula grooved, little palate movement seen, palate shaped in a ‘^’. No notch felt at hard palate border. The impression of submucous cleft palate was confirmed by the cleft surgeon and Maia was listed for surgery. Since she has slightly unusual facial features she was also offered screening for 22q11 deletion syndrome (see Chapter 12). Maia also needed speech therapy to show her how to make oral sounds. Following surgery this continued, to show her how to use her new palate structure.

g Further Reading: Peterson-Falzone, S., Trost-Cardamone, J., Karnell, M. and Hardin-Jones, M. (2006). The Clinician’s Guide to Treating Cleft Palate Speech. St. Louis: Elsevier Mosby. Sommerlad, B.C., Fenn, C., Harland, K., Sell, D., Birch., M.J., Dave R., Lees M. & Barnett, A. (2004). Submucous cleft palate: A grading system and review of 40 consecutive submucous cleft palate repairs. Cleft Palate-Craniofacial Journal 41(2), 114–123. Shprintzen, R.J. and Bardach, J. (1995). Cleft Speech Management: A Multidisciplinary Approach. St. Louis: Elsevier Mosby.

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t Useful Link: http://speech-language-therapy.com/Caroline Bowen

References Kummer, A. (2005). Ankyloglossia: To clip or not to clip? That’s the question. ASHA Leader, 10(17), 6–7, 30. Messner, A.H., Lalakea, M.L., Aby, J., Macmahon, J. & Bair, E. (2000). Ankyloglossia: Incidence and associated feeding difficulties. Archives of Otolaryngology – Head & Neck Surgery, 126(1), 36. Messner, A.H. & Lalakea, M.L. (2002). The effect of ankyloglossia on speech in children. Archives of Otolaryngology ­– Head & Neck Surgery, 127(6), 539–545. Stenglehofen, J. (1999). Working with Cleft Palate. Oxford: Winslow Press. Stewart, J.M., Ott, J.E. & Lagace, R. (1972). Submucous cleft palate: Prevalence in a school population. Cleft Palate Journal, 9, 246–250.

3 Section 2 Speech Assessment Ginette Phippen and Sandra Treslove

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

Aim of this section • To describe a framework for the assessment of speech in individuals with cleft palate and velopharyngeal dysfunction (VPD). • To provide quick reference guides for identifying and describing disorders of resonance, nasal airflow and cleft speech characteristics. • To consider how the impact of cleft speech disorder can be evaluated and how this contributes to the process of clinical decision-making.

Introduction Perceptual descriptions in speakers with cleft palate have generally included judgements of articulation, intelligibility, resonance and voice, as well as relationships with instrumental assessment of resonance (Kuehn & Moller, 2000). The Great Ormond Street Speech Assessment, GOS.SP.ASS (Sell, Harding & Grunwell, 1999) is the UK adopted framework for assessment of cleft palate speech. GOS.SP.ASS includes assessment of: • Resonance • Nasal emission and nasal turbulence • Consonant production and cleft speech characteristics (CSCs). Other areas observed and recorded include grimace, visual appearance of speech, oral examination and voice (see Figure 3.2.1).

• Note Language development Young children with cleft palate often have delayed language development, particularly in the area of expressive language (Persson et al., 2012; Russell & Harding, 2001). It is therefore important to include language screening alongside speech assessment in order to make appropriate decisions about management.

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The velopharyngeal mechanism in speech The velopharyngeal (VP) mechanism consisting of the velum (soft palate) and the pharynx (throat) has been extensively explored. Videoradiographic and endoscopic studies have confirmed that despite variability during speech, muscle activity causes the soft palate to elevate and retract which, along with

Figure 3.2.1 The GOS.SP.ASS (Continued over page)

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Speech Therapy in Cleft Palate and Velopharyngeal Dysfunction

(Continued from previous page)

(Sell, Harding & Grunwell, 1999). Reproduced by kind permission.

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movement of the muscles in the throat, results in the soft palate making a seal with the throat. This is described as velopharyngeal closure (Moon & Kuehn, 2004). This occurs for the majority of English speech sounds and serves to seal the mouth off from the nose in order to achieve the correct balance of oral and nasal resonance. Only for the sounds /m/, /n/ and /ŋ/ does the soft palate remain lowered to allow air to resonate in the nose. In some languages there may also be incomplete VP closure during the production of vowel sounds. Irrespective of accent, dialect or language it is clear that rapid, automatic and coordinated movement of the soft palate is needed during conversational speech to achieve the required balance of resonance (Bzoch, 2004).

Assessing resonance The term ‘nasal’ is commonly used to describe speech where there is too much or too little air resonating in the nasal cavity (Sell et al., 1999). In all speakers there is a balance of acoustic energy (sound) from air flowing from the lungs through the vocal cords into the cavities of the mouth and the nose (Jones, 2006). This is perceived by the listener as resonance, which is a quality of voice (Mathieson, 2001). There is a spectrum of resonance from hypernasality to hyponasality. If there is too much nasal resonance, caused by excess air passing through the nose, this is perceived as hypernasality. Conversely, a reduction in nasal resonance results in a hyponasal voice quality (Sweeney, 2011). Both hypernasality and hyponasality may be clinically significant as they can interfere with the clarity of speech. However, hyponasality is generally considered to have less of an impact on speech as it primarily affects only the nasal class of consonants in English speakers, i.e., /m, n, ŋ/. Hypernasality, on the other hand, affects all vowel sounds, can result in weak or substituted consonants (Harding & Grunwell, 1996) and has been reported as more negatively perceived by the listener (Lallh & Rochet, 2000).  Resource CD: The Rapid Assessment of Resonance (see below) has been devised as a quick reference guide for use by ENT professionals and community speech and language therapists. This is also included in photocopiable form on the Resource CD. This is intended as a guide to differential diagnosis and need for onward referral for investigation of suspected VPD.

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Rapid Assessment of Resonance Ask the child to say:

The child says:

Outcome/Action:

Mummy, mummy, mummy Nanny, nanny, nanny

Bubby, bubby, bubby Daddy, daddy, daddy

Hyponasality: adenoids, catarrh, nostrils not patent

Ask the child to say:

The child says:

Outcome/Action:

Daddy, daddy, daddy Bobby, bobby, bobby

Nanny, nanny, nanny Mommy, mommy, mommy

Hypernasality: Gently hold nose to see if it improves sound production. Look for signs of submucous cleft palate Refer to specialist cleft team

Ask the child to count:

You hear:

Outcome/Action:

four, five, six, seven

Snorting noises/escape of air down the nose with ‘f ’ or ‘s’ Other sounds are clear with no nasal air escape

Could be mislearned sounds (active nasal fricatives*) Often associated with middle ear hearing loss and nasal obstruction

*Active nasal fricatives are mislearned speech sounds where the child has learned to direct air out of the nose to produce a ‘snorting’ sound which replaces specific fricative sounds. To differentially diagnose, ask the child to say a long ‘sssssssssss’ and gently hold the nose. If holding the nose stops him/her producing any sound at all and you get the impression of the sound being ‘stuck somewhere’, then it is an active nasal fricative. If holding the nose helps him/her say a clear ‘s’, it could indicate some palatal dysfunction. *If active nasal fricatives please refer to your local SLT team (unless child has cleft palate/ submucous cleft too). If you suspect that a child has velopharyngeal dysfunction please refer to the local specialist cleft palate team. Devised by: The Spires Cleft Centre Speech and Language Therapy Service 2011

Nasal Airflow Abnormal nasal airflow accompanying oral consonants is described as nasal emission or nasal turbulence (see page 16). This can be confirmed by observing misting on a mirror held under the nostrils, as consonants are repeated. It is important to distinguish this from active nasal fricative (see Chapter 10.2).

Cleft speech characteristics (CSCs) As well as affecting resonance, cleft palate/VPD can also interfere with the development of speech sounds (Harding & Grunwell, 1996; Riski & DeLong, 1984). Substituted sounds may develop as a compensatory mechanism due to the early structural inadequacy of the VP mechanism, which prevents the required build-up of intraoral air pressure to form consonant sounds such as

Anterior CSCs

Posterior CSCs

Non-oral CSCs

Targets realised as lateral fricative [ɬ] [ɮ], lateralised fricatives [l͡s] [l͡z] or with lateral release e.g., [tˡ] N.B. In lateralised articulations, primary airstream is central, but air escapes simultaneously on one or both sides of the tongue

Typically alveolar plosives, fricatives, affricates /s z t ʧ ʃ / Less frequently velars

Typically alveolar plosives, fricatives, affricates /t d s z ʧ ʤ/ Less frequently velars

Usually bilabial or alveolar plosives

Bilabial, alveolar, velar consonants

Plosives, affricates and fricatives

Typically /s/ and /z/ but also /ʃ Ʒ ʧ ʤ/

Lateral articulation / lateralisation

Palatal articulation / palatalisation

Double articulation

Backing to velar, to uvular

Non-oral articulations

Active nasal fricatives

Sources: Grunwell & Sell (2001); Sell, Harding & Grunwell (1998); Sell, Harding & Grunwell (1994).

The target oral sound is replaced with a nasal fricative (see Chapter 10.2), with nasal emission, e.g., [n̥͋] or turbulence e.g., [ñ̥̃ ]

Pressure consonants replaced by glottal fricatives [h ɦ] pharyngeal fricatives [ħ ʕ] and/or glottal plosive /ʔ/. Often replacing voiceless plosives. Speaker compensates for reduced intra-oral pressure by attempting to articulate where more pressure can be achieved. N.B Word initial/final glottal plosives are easily misinterpreted as consonant deletion but may signal phonological contrast

e.g., Backing to velar [k g x ɣ] and uvular [q ɢ χ ʁ]

Articulation of two sounds simultaneously, most typically /t/ > [t͡k] and /d/ > [d͡g] Includes co-occurrence with glottal stops, e.g., /p/ >[p͡ʔ]

In palatalisation, the primary target is realised but with secondary approximation between tongue and hard palate. Heard as an /i/ or /j/ off-glide to the target, e.g., [tʲ]

Comments Often associated with class III malocclusion Dentalisation is a type of fronting; distinguish between fronting as a speech sound immaturity and as a CSC relating to structure

Cleft speech characteristic Frequently affected sounds Dentalisation/ Alveolar consonants imprecise tongue tip /t d n s z l/ movements

Active Cleft Speech Characteristics (CSCs)

Table 3.2.1 Summary of active cleft speech characteristics

Speech Assessment 49

Comments Air flows through nasal as well as oral cavity. Oral consonants weakened/nasalised due to reduced intraoral pressure, e.g., /b/ > [b ͉] or [b̃]

Oral consonants

Fricatives and plosives

Oral consonants

Fricatives/affricates

Weak, nasalised consonants

Nasal realisations

Absent pressure consonants

Gliding

Sources: Grunwell and Sell (2001), Sell, Harding and Grunwell (1998; 1994)

Targets realised as glides [w j ]

Speech contains only nasals and approximants, and/or glottal fricative /h/

Target sound replaced by its nasal equivalent, e.g., /b/ > [m], /d/ > [n] and /s/ > [(s)n̥] N.B: Nasal realisation of fricatives may be perceptually indistinguishable from active nasal fricatives (See Chapter 10.2 for differential diagnosis)

͜

Cleft speech characteristic Frequently affected sounds

Passive Cleft Speech Characteristics (CSCs)

Table 3.2.2 Summary of passive cleft speech characteristics

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/b/, /d/ and /g/. Russell and Harding (2001) explain how these articulation errors can become established at an early age in children with cleft palate resulting in a significant impact on speech clarity. CSCs can affect either or both place and manner of production, for example backing of alveolar to velar consonants or nasal realisation of plosive consonants. They are categorised as either active or passive. Table 3.2.1 describes the substitutions classified as active CSCs. The term active refers to articulations that are actively produced as alternatives to target consonants (Harding & Grunwell, 1998). These are further categorised according to whether the substitution is anterior, posterior or non-oral. Table 3.2.2 describes the substitutions classified as passive CSCs. The term passive refers to realisations that arise from VPD and/or fistulae. The potential to achieve and/or sustain intraoral pressure is reduced, hence the term ‘passive’ (Harding & Grunwell, 1998). g Further Reading: Sell, D. and Grunwell, P. (2001). Speech assessment and therapy. In: A. Watson, D. Sell and P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Grunwell, P. (Ed.) (1993). Analysing Cleft Palate Speech. London: Whurr Publishers.

Transcription Transcription of disordered speech is challenging and requires continued practice to maintain skills. A set of Good Practice Guidelines has been issued by the Specialists in Specific Speech Impairment Network (2012) which outlines the importance of transcription for: • Differential diagnosis • Establishing the nature and extent of the speech difficulty • Management and service delivery decisions • Selection of intervention approach • Measuring change. The guidelines propose that SLTs need to be confident in using the full set of IPA (International Phonetic Alphabet) and if necessary ExtIPA (Extended International Phonetic Alphabet) symbols ‘where phonetic-level variation from standard speech is likely’. In practice, it is perfectly acceptable to refer to the IPA/ExtIPA charts after live transcription where there is uncertainty

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about the correct symbol(s). In these cases it is preferable for the SLT to try to describe the nature of the perceived sound rather than omit transcription altogether, or transcribe according to the expected target sound. In complex cases, including some children with cleft palate, the use of video-recording is recommended to allow for review of the speech sample. Examples of common CSC symbols and diacritics are shown in Tables 3.2.1 and 3.2.2 and listed for reference below: Dental

[d̪]

Lateral/ised

[ɬ] [ɮ] [tˡ]

Palatal/ised

[c] [ç] [tʲ]

Double articulation

[t͡k] [p͡ʔ]

Uvular

[q ɢ χ ʁ]

Glottal

[h ɦ]

Pharyngeal

[ħ ʕ]

Active nasal fricative

[n̥]͋ [ñ̥]̃

Weak/nasalised

[b ͉] [b]̃

Nasal emission

[t]

Nasal turbulence

[s]

Evaluating impact The impact of cleft palate and VPD As previously discussed, children born with cleft palate are at risk of developing compensatory articulatory patterns known as cleft speech characteristics. These patterns can quickly become established as part of the child’s contrastive phonological system and may be resistant to therapy in the presence of persisting VPD. The primary impact of VPD (cleft and non-cleft) is on speech, although nasal regurgitation of food and drink, and nasal or facial grimacing can also be features (Bradbury, 2001). A nasal speech quality sounds noticeably abnormal to the listener, making speech weak, indistinct and difficult to understand. The substitution of nasal sounds for their oral counterparts, so that /b/ is perceived as /m/, for example, significantly reduces speech clarity so that the word ‘baby’ sounds like ‘mamy’ and ‘daddy’ sounds like ‘nanny’. Similarly,

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the noisy escape of air accompanying speech (nasal turbulence) is intrusive in conversation (Wyatt et al., 1996).

A word on severity The severity of nasal speech and/or CSCs associated with cleft palate/VPD varies between individuals, with a mildly nasal voice quality at one end of the spectrum and severely unintelligible speech at the other. The terms mild, moderate and severe are widely used in healthcare to indicate symptom severity. These descriptive terms have no absolute values although health researchers have attempted to clarify and quantify the boundaries between them, for example, in defining degrees of pain (Serlin et al., 1995). The terms are also seen in definitions of severity and levels of impairment in speech disorders (ASHA, 2010; Bowen, 1998). In relation to nasal speech these relative descriptors are also widely used in speech assessment following cleft palate repair (Vogel et al., 2009; Henriksson et al., 2005; Lee et al., 2003). However, as discussed in more detail in Chapter 1, it appears that the degree of impact on communicative confidence and self-esteem is not necessarily correlated with the severity of the communication impairment (McLeod, 2004). It has been postulated that this variability may relate to psychological resilience whereby people differ in their ability to cope with negative events, including health difficulties, depending on individual traits as well as the influence of external factors (Tugade et al., 2004).

Why does impact matter? The impact of speech, language and communication needs is well documented (Markham et al., 2009). A systematic review of relevant papers over a period of 10 years by McCormack et al. (2009) revealed that speech impairment in childhood may be associated with limitations or restrictions in a wide range of domains including reading, writing, thinking, relationships and work. There are reports of bullying (Knox & Conti-Ramsden, 2003), withdrawal (Fujiki et al., 2001) and poor academic achievement (Lewis et al., 2004). Children whose impairments involve speech alone are generally thought to be less at risk than those whose impairments involve language, particularly understanding what others say (Young et al., 2002; Law et al., 1998). However, Lewis and Freebairn (1992) also found evidence that early difficulties with phonology (speech sounds) are associated with poor performance in reading and spelling in adolescents and adults. Similarly, children with voice disorders may be perceived more negatively than children with normal voice (Hooper, 2004).

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Assessing impact The question of how to assess impact is becoming more widely considered and various tools have emerged to evaluate the effect of communication difficulties on quality of life in children and adults (McLeod, 2004). The importance of impact assessment is clear as there are arguably two distinct skills in the clinical decision-making process: first of all, the ability to systematically apply diagnostic and research information and, secondly, to consider the contextual factors and their impact (Weiner et al., 2007). Assessment is the process by which information is collected from a variety of sources, often within a specific framework, depending on the clinical context. This forms the basis for analysis of the information and choice between alternatives, i.e., clinical judgement (Hardy & Smith, 2008). Assessment is more than just data; it involves the interpretation of cues that include evidence, opinion and experience. This is particularly true when assessing the impact of speech and language difficulties in general (Markham et al., 2009). Whichever tool is selected, informal or formal, standardised or not, there is a need to explicitly ask the ‘impact question’, i.e., how is the child or adult affected by their cleft-related speech difficulties or VPD? The answer may be obtained from the individual themselves, parents, teachers or other professionals, and most likely from a combination of all of these. This will inform the decision-making process for therapy intervention and in some cases surgery, highlighting the importance of this aspect of the assessment process.

References American Speech & Hearing Association (ASHA) (2010). Motor Speech Disorders Evaluation. [online] Available at: http://www.asha.org/uploadedFiles/slp/healthcare/AATMotorSpeech. pdf [Accessed 11 October, 2010] Bowen, C. (1998). Children’s speech sound disorders: Questions and answers. [online] Available at: http://www.speech-language-therapy.com/phonol-and-artic.htm [Accessed 11 September, 2010] Bradbury, E. (2001). Growing up with a cleft: The impact on the child. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Bzoch, K.R. (2004). Introduction to the study of communicative disorders in cleft palate and related craniofacial anomalies. In K.R. Bzoch (Ed.), Communication Disorders Related to Cleft Lip and Palate. Austin, Texas: Pro-Ed.

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Fujiki, M., Brinton, B., Isaacson, T. & Summers, C. (2001). Social behaviors of children with language impairment on the playground: A pilot study. Language Speech & Hearing Services in Schools 32(2): 101–113. Good Practice Guidelines for Transcription of Children’s Speech Samples in Clinical Practice and Research (2012). Specialists in Specific Speech Impairment Network, circulated by email January 2013. Harding, A. & Grunwell, P. (1996). Characteristics of cleft palate speech. European Journal of Disorders of Communication 31(4), 331–357. Harding, A. & Grunwell, P. (1998). Active versus passive cleft-type characteristics: Implications for surgery and therapy. International Journal of Language and Communication Disorders 33(3), 329–352. Hardy, D. & Smith, B. (2008). Decision making in clinical practice. British Journal of Anaesthetic and Recovery Nursing, 9(1), 19. Henriksson, T.G., Hakelius, M., Andlin-Sobocka, A., Svanholm, H., Low, A. & Skoog, V. (2005). Intravelar veloplasty reinforced with palatopharyngeal muscle: A review of a 10year consecutive series. Scandinavian Journal of Plastic and Reconstructive Surgery and Hand Surgery, 39(5), 277–282. Hooper, C.R. (2004). Treatment of voice disorders in children. Language Speech & Hearing Services in Schools, 35(4), 320–326. Jones, D.L. (2006). Patterns of oral-nasal balance in normal speakers with and without cleft palate. Folia Phoniatrica et Logopaedica, 58, 383–391. Knox, E. & Conti-Ramsden, G. (2003). Bullying risks of 11-year-old children with specific language impairment (SLI): Does school placement matter? International Journal of Language & Communication Disorders, 38(1), 1–12. Kuehn, D.P. & Moller, K.T. (2000). Speech and language issues in the cleft palate population: The state of the art. The Cleft Palate-Craniofacial Journal, 37(4), 348–350. Lallh, A.K. & Rochet, A.P. (2000). The effect of information on listeners’ attitudes toward speakers with voice or resonance disorders. Journal of Speech Language & Hearing Research, 43(3), 782–795. Law, J., Boyle, J., Harris, F. & Harkness, A. (1998). Screening for primary speech and language delay: A systematic review of the literature. International Journal of Language & Communication Disorders 33(S1), 21–23. Lee, A.S.Y., Ciocca, V. & Whitehill, T.L. (2003). Acoustic correlates of hypernasality. Clinical Linguistics & Phonetics, 17(4–5), 259–264. Lewis, B.A. & Freebairn, L. (1992). Residual effects of preschool phonology disorders in grade school, adolescence, and adulthood. Journal of Speech & Hearing Research, 35(4), 819–831. Lewis, B.A., Freebairn, L.A., Hansen A.J., Iyengar, S.K. & Taylor, H.G. (2004). School-age follow-up of children with childhood apraxia of speech. Language Speech & Hearing Services in Schools, 35(2), 122–140.

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McCormack, J., McLeod, S., McAllister, L. & Harrison, L.J. (2009). A systematic review of the association between childhood speech impairment and participation across the lifespan. International Journal of Speech-Language Pathology, 11(2), 155–170. McLeod, S. (2004). Speech pathologists’ application of the ICF to children with speech impairment. Advances in Speech-Language Pathology, 6(1), 75–81. Markham, C., Van Laar, D., Gibbard, D. & Dean, T. (2009). Children with speech, language and communication needs: Their perceptions of their quality of life. International Journal of Language & Communication Disorders, 44(5), 748–768. Mathieson, L. (2001). In M.L.C. Greene & L. Mathieson (Eds), The Voice and its Disorders, 6th Edn. London: Whurr Publishers. Moon, J.B. & Kuehn, D.P. (2004). Anatomy and physiology of normal and disordered velopharyngeal function for speech. In K.R. Bzoch (Ed.), Communicative Disorders Related to Cleft Lip and Palate. Austin, Texas: Pro-Ed. Persson, M., Becker, M. & Svensson, H. (2012). Academic achievement in individuals with cleft: A population-based study. Cleft Palate-Craniofacial Journal, 49(2), 153–159. Riski, J.E. & DeLong, E. (1984). Articulation development in children with cleft lip/palate. Cleft Palate Journal, 21(2), 57–64. Russell, J. & Harding, A. (2001). Speech development and early intervention. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Sell, D., Harding, A. & Grunwell, P. (1994). GOS.SP.ASS. A screening assessment of cleft palate speech. European Journal of Disorders of Communication, 29(1), 1–15. Sell, D.A., Harding, A. & Grunwell, P. (1999). GOS.SP.ASS.’98: An assessment for speech disorders associated with cleft palate and/or velopharyngeal dysfunction (revised). International Journal of Language & Communication Disorders, 34(1), 17–33. Sell, D.A. & Grunwell, P. (2001). Speech assessment and therapy. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Serlin, R.C., Mendoza, T.R., Nakamura, Y., Edwards, K.R. & Cleeland, C.S. (1995). When is cancer pain mild, moderate or severe? Grading pain severity by its interference with function. Pain, 61(2), 277–284. Sweeney, T. (2011). Nasality – Assessment and intervention. In S. Howard & A. Lohmander (Eds), Cleft Palate Speech: Assessment and Intervention. Chichester, UK: Wiley-Blackwell. Tugade, M.M., Fredrickson, B.L. & Feldman Barrett, L. (2004). Psychological resilience and positive emotional eranularity: Examining the benefits of positive emotions on coping and health. Journal of Personality, 72(6), 1161–1190. Vogel, A.P., Ibrahim, H.M., Reilly, S. & Kilpatrick, N. (2009). A comparative study of two acoustic measures of hypernasality. Journal of Speech Language & Hearing Research, 52(6), 1640–1651.

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Weiner, S.J., Schwartz, A., Yudkowsky, R., Schiff, G.D., Weaver, F.M., Goldberg, J. & Weiss, K.B. (2007). Evaluating physician performance at individualizing care: A pilot study tracking contextual errors in medical decision making. Medical Decision Making, 27(6), 726–734. Wyatt, R., Sell, D., Russell, J., Harding, A., Harland, K. & Albery, E. (1996). Cleft palate speech dissected: A review of current knowledge and analysis. British Journal of Plastic Surgery, 49(3), 143–149. Young, A.R., Beitchman, J.H., Johnson, C., Douglas, L., Atkinson, L., Escobar, M. & Wilson, B. (2002). Young adult academic outcomes in a longitudinal sample of early identified language impaired and control children. Journal of Child Psychology and Psychiatry, 43(5), 635–645.

g Further Reading: Grunwell, P. (Ed.) (1993). Analysing Cleft Palate Speech. London: Whurr Publishers. Sell, D. & Grunwell, P. (2001). Speech assessment and therapy. In A. Watson, D. Sell and P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers.

4 Assessment and Management of Velopharyngeal Dysfunction (VPD) Leda Koutsoulieri

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Aim of this chapter This chapter explores the instrumental procedures used for assessment of VPD as well as the different management options when VPD is confirmed.

Introduction Assessment of velopharyngeal dysfunction (VPD) usually takes place in specialist clinics, often called palate investigation clinics, attended by a consultant plastic surgeon and a specialist speech and language therapist. These clinics are designed for individuals who have suspected VPD and the aim is to confirm or exclude VPD, investigate its cause and extent and decide on the appropriate management option.

Instrumental evaluation of VPD Why do we need instrumental assessment? Perceptual speech evaluation is at the centre of VPD assessment, as it is the listener’s subjective judgement that determines whether an individual’s speech is considered atypical (Sell & Grunwell, 2001). For many years the trained clinician’s ear was the only tool available to assess the speech of an individual with cleft palate or VPD. Although the well-trained ear can detect a speech disorder, it cannot describe the anatomy causing the problem (Muntz, 1992). Objective measures such as instrumentation to directly visualise the velopharyngeal (VP) mechanism are now routinely used alongside perceptual speech assessment. This allows the clinician to better understand the structure and function of the VP mechanism by establishing its status: essential for any physical management of the defect. Furthermore, the outcome of any intervention can be visualised and documented. Instrumentation can also act as a ‘back up’ to clinicians’ perceptual assessment, particularly in cases of borderline or inconsistent VPD, where objective assessment can tip the balance of clinical decision making either toward or away from surgical intervention (Peterson-Falzone et al., 2006).

Types of instrumental assessment The gold standard for the assessment of velopharyngeal status consists of

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two direct visualisation procedures: videofluoroscopy and nasendoscopy (Shprintzen, 1995).

• Terminology

Fluoroscopy is an imaging technique that uses X-rays to obtain real-time moving images of the internal structures of the body. This is achieved by a patient being positioned between a source that transmits X-rays through the body, and a digital detector that absorbs the X-rays exiting the body. The amount of X-rays exiting the body will depend on the density of the body tissues it is passing through, which is how different structures are defined on an image. The X-rays that exit the body are then converted into a signal that is transmitted as an image onto a television monitor.Videofluoroscopy is when the real-time images are recorded for future playback as real-time images rather than stationary images. Endoscopy means looking inside and typically refers to the use of an instrument that can be passed through body openings and pathways to examine internal organs or cavities of the body. Nasendoscopy refers to an endoscope passed through the nasal cavities. It allows for examination of the anatomy and physiology of the velopharynx during speech. A recent survey of the 16 cleft centre sites in the UK showed that all routinely use videofluoroscopy and 70% use nasendoscopy. However, international practice varies; for example, in the US, nasendoscopy is often the investigation of choice due to issues such as radiation exposure and difficulties in interpreting videofluoroscopy (Kummer et al., 2012; Sell & Pereira, 2011). Despite this, it is widely accepted that both videofluoroscopy and nasendoscopy are important, as each provides unique but complementary information.

Videofluoroscopy There are a number of angles that can be used to visualise the VP mechanism with the following views most commonly described in the literature (PetersonFalzone et al., 2010; Shprintzen, 1995): Base view: described as an ‘en face’ view of the velopharyngeal mechanism. The X-ray beam is oriented vertically through the pharynx by placing the patient on the fluoroscopic table in a ‘sphinx-

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like’ position with head hyper-extended backwards. This view has some drawbacks (such as positioning difficulties). Furthermore, information gained from this view can also be seen in nasendoscopy, which, it has been argued, provides a better ‘en face’ view. Frontal view: Obtained with the individual in supine position as the image intensifier is passed directly over the face (so that the patient is facing it). Frontal views provide information about lateral pharyngeal wall movement but do not clearly show the relationship of lateral pharyngeal walls to the soft palate. Towne’s view: This view has the individual either sitting upright or supine with the head flexed downward (chin close to chest). The X-ray beam is perpendicular to the VP port. This view has been described as similar to the base view. Again, nasendoscopic views are comparable and may be preferred to avoid extra radiation exposure. Lateral view: To obtain this view, the individual stands or sits with the midsagittal plane perpendicular to the X-ray beam (side view). The soft palate, posterior pharyngeal wall and tongue are easily seen in this position. It is suggested that the following information is required to fully assess the anatomy and function of the VP mechanism using videofluoroscopy (Sell & Pereira, 2011; Shprintzen, 1995): • Soft palate movement and structure • Posterior pharyngeal wall movement and structure • Tongue movement, position and interaction with soft palate during articulation (e.g., tongue humping to assist VP closure) • Firmness of velopharyngeal closure and plane of closure against the posterior pharyngeal wall • Adenoids and tonsils (size and location) • Position of levator palatini muscles (e.g., anterior or not) • Presence of Passavant’s ridge and role in VP closure. In order to observe the above, frontal and lateral views have been suggested as most useful and other views would only be complementary. The lateral videofluoroscopy view is most commonly used because it allows the clinician

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to make the above observations. Information shown in frontal view but not lateral view, such as aspects of the lateral pharyngeal wall movements, can be provided by nasendoscopy, so the frontal view is not routinely employed.

• Terminology

Passavant’s ridge: defined as a localised anterior projection from the posterior pharyngeal wall (back of throat) that is not permanent but appears during certain velopharyngeal valving activities, such as speech. There is some controversy as to which muscle is responsible for forming this ridge as well as whether it actually assists velopharyngeal closure (PetersonFalzone et al., 2010). Tongue humping: refers to the use of the tongue in an attempt to assist velopharyngeal closure. Some individuals with VPD may use the back of their tongue to lift the soft palate during speech. This can be observed in lateral videofluoroscopy but not nasendoscopy, as during this latter view the tongue is below the palate and therefore hidden from view (Shprintzen,1995). Speech sample The speech sample during videofluoroscopy usually includes consonant-vowel sequences (e.g., “pa-pa, pi-pi”), sentence repetition and rote speech such as counting from 1–20. However, the sample is often tailored to the individual; for example, a young child with a limited consonant repertoire may be asked to only produce or copy the consonant(s) he/she is able to make.

Nasendoscopy Nasendoscopy is an imaging procedure in which a small flexible fibreoptic endoscope is inserted into one of the individual’s nasal passages for direct viewing of the VP mechanism from above. The use of this procedure for the investigation of VPD was first described by Pigott and colleagues in 1969 (Mercer & Pigott, 2001). The endoscope consists of a viewing lens, a fibreoptic insertion tube, an eyepiece attached to a camera and a high-intensity light source. Videorecording equipment is also needed where both the clinician and patient can see the images (Peterson-Falzone et al., 2010). Passing the endoscope through the nose does not interfere with the normal production of speech while at the same time it provides a direct view of the VP mechanism.

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Nasendoscopy can on occasions be too invasive for some small children and indeed adolescents and adults. Application of topical anaesthesia is recommended, as well as leaving enough time between the application and the performing of the procedure to ensure that the anaesthetic is fully effective. Explaining the procedure to children in a way they can understand is important, as well as telling them they will be able to feel it but that it does not hurt. An age-appropriate testing environment and clinician manner, as well as family support, will contribute to a high success rate. The most advantageous viewing angle is achieved by passing the scope through the middle meatus of the nose (Shprinzten, 1995; Muntz, 1992) although the inferior meatus is often used. Nasendoscopy allows for direct

Figure 4.1 Videofluoroscopy equipment

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visualisation of both the anatomical structures and functional features of the VP mechanism, outlined below (Sell & Pereira, 2011). Structural: • The nasal surface of the soft palate • Presence/size of adenoids • Abnormal position of carotid arteries (vascular pulsations) • Abnormal growths and structures • Anatomy of the posterior pharyngeal wall.

Figure 4.2 Nasendoscopy equipment

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• Note:

Position of the major neck vessels is important to establish, particularly in cases where VP management involves a pharyngoplasty operation. In some individuals with 22q11 deletion syndrome, there may be abnormal medial displacement of the internal carotid arteries. A sign of this is vascular pulsations in the throat observed during endoscopy, showing that the arteries are displaced too close to the surface, or that the pharyngeal walls are thinner than normal. Due to the risk of damaging the internal carotid artery during surgery, the surgeons often prefer to establish the position of this artery pre-operatively, usually with an MRI scan (Shprintzen, 1995).

Functional: • Velar movement and symmetry of movement • Synergistic movement of velum, posterior pharyngeal wall and lateral pharyngeal wall • VP closure pattern (e.g., circular, sagittal, coronal) • Presence of VP gap, including size, symmetry and location. Speech sample The elicited speech sample again usually involves spontaneous and rote speech, imitation of sentences and consonant-vowel syllables and/or, as in the case of the videofluoroscopy sample, any consonant sounds that the child is able to produce/copy. Both videofluoroscopy and nasendoscopy procedures are recorded for future playback purposes. g Further Reading: Sell, D. et al. (2008) Speech and language therapy and nasendoscopy for patients with velopharyngeal dysfunction. RCSLT Position Paper. Royal College of Speech and Language Therapists, London, UK

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Magnetic Resonance Imaging (MRI) MRI scans have been used to visualise the VP mechanism, as they are well suited to imaging internal soft tissues. MRI is based on the principles of electromagnetic energy and therefore lacks the risks associated with radiographic techniques (X-rays). Among its advantages are its non-invasive nature, its repeatability and reproducibility, as well as allowing visualisation of the underlying soft palate muscles with good soft tissue contrast and high spatial resolution. However, it is not widely used due to the limitation of providing still images of static soft tissue rather than dynamic speech. In addition, expense, limited availability and complexity of the procedure all make this approach impractical for routine clinical use (Sell and Pereira, 2011; Peterson-Falzone et al., 2010).

Summary In summary, lateral videofluoroscopy and nasendoscopy are the gold standard for instrumental assessment of the VP mechanism. Shprintzen (1995) emphasises the need for using both procedures, suggesting that nasendoscopy is best for observing structural aspects of the VP mechanism, while videofluoroscopy provides better information regarding its function. A study by Lipira et al. (2011) reported a moderate correlation between nasendoscopy and lateral videofluoroscopy VP closure estimates. Interestingly, videofluoroscopy estimates tended to underestimate gap size, which the authors suggested happens because in this procedure the defect is visible if it spans the entire width of the VP space so that in cases of asymmetric closure or a midline gap the magnitude of the defect is not shown.

Other types of instrumental assessment Acoustic measurements: Nasometry The Nasometer™ is a computer-based instrument that computes a ratio between acoustic energy (detected by microphones) at the nose level and mouth level during speech (Figure 4.3). The resulting score is termed ‘Nasalance’ and is calculated by dividing the intensity of nasal to nasal-plus-oral acoustic energy during speech. The Nasometer™ consists of a headset with a sound separator between the nose and mouth with microphones on both sides, a Nasometer™ unit, computer software and a calibration unit. The sound signal is picked up by the microphones, is filtered and digitised and then processed by the computer.

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Figure 4.3 Picture of Nasometer™

The data are provided in both a graphic representation and a quantitative percentage result (Sweeney, 2011). Nasalance measurements can range from near 0 (very little) to 100 (excessive) (Peterson-Falzone et al., 2006). Normal scores for a standard speech sample can range from 25% to 32% although there are regional variations (Sell & Grunwell, 2001). Factors such as language, dialect and speech stimuli used can all influence scores. The Nasometer™ provides an instrumental quantification of nasality and therefore can be used in the assessment of VPD, both before and after treatment, to supplement perceptual ratings. Nasometry can also be used in therapy as a visual biofeedback tool (see Chapter 10.4). Aerodynamic measurements These refer to devices that measure nasal airflow and oral air pressure during sound production and provide information about the coupling of the oral and nasal cavities during speech. One such system is PERCI-SARS. Pressure differences on either side of the VP port are measured using nasal and oral pressure transducers and an estimate of the VP port area is calculated by use of the differential pressure and nasal airflow (Sell & Grunwell, 2001). Aerodynamic measurements are indirect measures of VP function, because

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Figure 4.4 VPD management flow chart

they do not allow for direct visualisation of the VP structures. They are useful for research purposes but not commonly used in clinical practice, as they can be complex to use in order to produce meaningful findings (Peterson-Falzone et al., 2010).

Management of VPD Management of VPD includes both surgical and non-surgical options (see Figure 4.4) and decisions are based on both perceptual and instrumental assessment procedures. Management is tailored to the individual, taking into

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consideration the structural and functional aspects of the VP mechanism, age, additional diagnoses, previous management, impact of VPD and last, but not least, the wishes of the individual and his/her family.

Surgical management of VPD (with or without SLT) Surgery is usually indicated if VPD is consistent and VP closure is not achievable (Sommerlad, 2002). Figures vary but it is generally reported that up to 25% of individuals who have received primary cleft palate surgery will require a subsequent surgical procedure to treat VPD (Kuehn and Moller, 2000). In addition, a number of individuals presenting with non-cleft VPD may benefit from a surgical procedure to improve VP function. There are different surgical approaches to VP management including: 1. Techniques to alter the muscle arrangement within the palate and improve its function – palatal surgery. 2. Techniques that do not change the palate itself but alter the size and shape of the VP mechanism to assist the palate in achieving closure against the pharyngeal wall – pharyngoplasty.

Palatal surgery This category of surgical procedures on the palate includes both surgery on the palate musculature (e.g., Sommerlad radical palate muscle retropositioning) and palatal lengthening procedures (e.g., Furlow double opposing Z-plasty, Buccinator flaps). Surgery on the palate may be indicated following inadequate repositioning of the levator muscle during primary cleft repair, or in an unrepaired submucous cleft palate. Palate re-repair is theoretically the most physiological operation as it involves attempting to restore the palate to a normal state and does not interfere with the pharynx (Mercer and Pigott, 2001). Procedures that increase palatal length should benefit individuals with good soft palate movement but where there is inadequate length to achieve closure against the pharyngeal wall (Peterson-Falzone et al., 2010).

Pharyngoplasty This second group of surgical procedures is indicated for individuals where surgery on the palate may not be appropriate, i.e., in the presence of severe palatal muscle deficiency or where VPD is not attributed to a cleft (see non-cleft VPD

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section, Chapter 2). In these cases where palate function cannot be improved, there are surgical techniques that alter the structure of the nasopharynx, to reduce its size and make VP closure easier. These operations principally involve: (a) midline pharyngeal flaps or (b) sphincter pharyngoplasties (Mercer and Pigott, 2001) with (c) injection augmentation occasionally used. Airway obstruction and/ or hyponasality may occasionally be undesirable outcomes of this type of surgery, so post-operative evaluation of both airway and speech is necessary. (a) Midline pharyngeal flap These flaps are of muscle and mucosa from the posterior pharyngeal wall that are raised and attached to the soft palate forming a bridge. Flaps can be superiorly based or inferiorly based and are inset in a variety of methods. With sufficient lateral pharyngeal wall movement they can allow for complete closure of the VP space. However, if there is not enough lateral pharyngeal wall movement, there may still be remaining gaps on either or both sides of the flap, leaving incomplete closure (Mercer and Pigott, 2001). To be successful in correcting VPD, pharyngeal flap surgery will depend on adequate flap width and height and lateral VP port size; a flap that is based too high/too low, is very narrow, or where there is not enough lateral pharyngeal wall movement to close the lateral ports may not result in successful elimination of VPD (Riski, 2011). Conversely, if a flap is too wide or occlusive, hyponasality may occur and even obstruction of the airway, both of which are unsatisfactory outcomes. Bardach (1995) argued that secondary pharyngeal surgery, particularly pharyngeal flaps, are not physiologic procedures as they create an anatomy that normally does not exist. It is therefore important to carefully consider this as an option, following detailed investigations of the VP mechanism. (b) Sphincter pharyngoplasties These surgical techniques aim to reduce the size of the VP space by transposing muscular tissue to form a ridge on the posterior pharyngeal wall, thus bringing the latter forward to assist closure with the soft palate (also referred to as augmentation pharyngoplasties). Hynes was among the first to attempt this muscle transposition and created such a ridge in the posterior pharyngeal wall by changing the position and orientation of some of the pharyngeal muscles (Hynes pharyngoplasty) (Peterson-Falzone et al., 2006). The Hynes procedure does not attempt to make a dynamic sphincter. The Orticochea pharyngoplasty is another variation of sphincter pharyngoplasty; it differs from the Hynes procedure in aiming to make a dynamic contracting sphincter behind the

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palate, which will contract in normal speech and assist VP closure (Orticochea, 1983). Surgeons have been modifying the sphincter pharyngoplasty by tailoring the height of flap insertion, and success rates have been shown to be higher when the flaps are placed at the level of active velar elevation (Riski, 2011). Failed pharyngoplasty procedures have been attributed to factors such as low flap insertion, flap dehiscence, flaps that are not approximated in midline, a VP gap that is too large to be treated with this procedure, and coexisting speech disorders (Riski, 2011). When planning a pharyngoplasty operation it is important to consider the size and position of the adenoids. Since Hynes flaps need to be positioned high in the adenoidal area to be effective, if the adenoid bulge is of a considerable size an adenoidectomy may be necessary prior to the pharyngoplasty (Goodacre, 2013). Patient selection for each type of speech surgery is of great importance, as the existing anatomical and functional characteristics of the velopharynx will determine which procedure is best for the individual’s needs. In some cases a combined procedure may be used, for example a palate re-repair together with a Hynes pharyngoplasty, based on evidence that either procedure alone is unlikely to result in significantly improved VP function. These operations can take place either in one procedure or separately, with palate re-repair usually taking place before pharyngoplasty (Goodacre, 2013). (c) Augmentation procedures by implants/injections This refers to augmentation of the posterior pharyngeal wall using autogenous or exogenous implants and injections. The aim is to create a bulge on the posterior pharyngeal wall so that the soft palate has less distance to travel to close the VP space during speech. When this is the procedure of choice, it is usually applied in small VP defects (Riski, 2011). Cartilage implants (from the rib) have been used in the past, as well as fat injections and synthetic materials (Teflon, Proplast and collagen, among others). However, a number of studies have shown limited success with these techniques due to unpredictable results, post-operative complications, restrictions imposed with regards to safe use of some synthetic materials, as well as problems with implants holding their position or size over time (Peterson-Falzone et al., 2010). These procedures therefore have limited use, although they are still offered to a selected number of patients.

Speech and language therapy input Surgical management of VPD is indicated in cases of consistent VPD. If the

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only pre-existing speech problem is hypernasality and/or passive nasal airflow errors, then an operation may have the desirable outcome of normalising speech. However, VPD can co-exist with other speech and language difficulties such as: • Cleft speech characteristics (CSCs) • Articulation/phonological difficulties unrelated to cleft/VPD • Language delay/disorder • Voice problems. The above difficulties will require SLT input, as surgery alone will not fully resolve the speech difficulties (Peterson-Falzone et al., 2006). In cases of severe VPD, SLT input for CSCs articulation problems may need to wait until after surgery, as an improved VP structure and ability to build up intraoral pressure can help speech sound production.

Pre-operative SLT input SLT input for articulation errors (CSCs) before surgery is advisable, particularly in cases that need secondary speech surgery such as a pharyngeal flap, on the basis that improved articulation may reduce the extent of the flap (Sell & Grunwell, 2001). For example, a child presenting with non-oral articulation (i.e., pharyngeal, glottal, active nasal fricatives) may benefit from speech therapy for production of oral sounds, resulting in improved palate function. In such cases, a 3–6 month period of diagnostic therapy is advocated, together with careful documentation of change. This is less relevant in cases of surgery on the palate (e.g., palate re-repair) and consistent VPD, as the timing of this type of surgery will not be affected by articulation improvement (Sell & Grunwell, 2001). Speech therapy input may also be advisable prior to perceptual and instrumental assessment of VP function, for example in cases where a child’s speech repertoire consists only of sounds that do not require VP closure (e.g., glottal and pharyngeal sounds or active nasal fricatives) (Mercer & Pigott, 2001). In these cases, palate function potential may not be clear in the absence of oral pressure consonants. Successful therapy to improve oral articulation will assist

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investigations of palate function during speech which will ultimately assist the decision making process of whether surgery is appropriate or not.

Non-surgical management of VPD Management of VPD does not always involve a surgical procedure, although it is a common treatment option for consistent VPD. There are cases, however, where surgery is not an option. Contraindications for surgery can be (Shifman et al., 2000): • Advanced cardiovascular or neurologic diseases • Biomechanical limitations for surgery (e.g., limited mandibular opening or microstomia) • Deviation of the internal carotid arteries as in cases of 22q11deletion syndrome • Other medical contraindications for surgery • Presence or risk of obstructive sleep apnoea • Previously failed surgeries • Patients declining surgery. For individuals who are not candidates for surgery, there are other management options. However, careful patient selection is important, as these options can present with challenges of their own, and may not be appropriate for all.

Prosthetic management of VPD Where surgery is contraindicated, speech prosthesis may be the treatment of choice. The purpose of a prosthetic appliance is to create a separation between the nasopharynx and oropharynx when VPD is present, thus obturating the VP port during speech. This can result in the elimination of hypernasality and passive nasal airflow errors and could provide the speaker with the ability to achieve normal intraoral pressure when other aspects of articulation and phonation are also normal (Golding-Kushner et al., 1995). Indications for prosthetic treatment include: previously failed surgeries; medical contraindications for surgery; no soft palate/severe tissue deficiency/large anatomical defect. Prosthetic appliances have also been used to temporarily facilitate SLT treatment for non-oral errors as well as to evaluate the potential benefits of surgery in some cases (Sell et al., 2006).

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There are a number of factors to be considered when selecting candidates for prosthetic treatment (Sell et al., 2006): • Aetiology and characteristics of the velopharyngeal defect • Age and developmental level • Associated syndrome • Patient and family motivation and commitment • Speech presentation. Careful consideration of patients’ characteristics and needs is important; Mazaheri (1996) and Gardner and Parr (1996) argue that contraindications for a prosthetic appliance exist, and include feasibility of surgical repair, patients not capable of caring for the appliance, patients not cooperative or motivated for this type of treatment, uncontrolled dental caries and lack of trained/ experienced professionals to construct the speech prosthesis. Golding-Kushner et al. (1995) also state that prostheses are of limited use in severely dysarthric patients in whom articulation is unintelligible even with nasal occlusion, as well as in cases of non-functional or severely impaired language skills. As a speech prosthesis may be the only or final option for some individuals, the timing of this intervention is paramount. For children in particular, it is better to wait until they are able to understand what the prosthetic appliance does and why it is important to wear it and look after it. Factors that have been associated with failure include young age, associated syndrome and low motivation for wearing the appliance (Sell et al., 2006). Prior to patient selection, it is important that a comprehensive assessment of velopharyngeal function takes place, as previously described. Multidisciplinary discussion with the plastic surgeon, speech and language therapist, orthodontist/restorative dentist as well as the patient and family will determine whether a prosthetic intervention is appropriate.

Types of prosthetic appliances (a) Palatal lift A palatal lift may be useful when VPD is due to inadequate velar elevation but the palate is of sufficient length and thickness to contact the posterior pharyngeal wall when elevated. This type of VPD usually occurs in an anatomically intact velopharynx that does not function well, such as in individuals with disorders

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Figure 4.5 Palatal lift Reprinted with kind permission from Cleft Palate Speech, 2010. Copyright Elsevier

of neurogenic origin. These patient groups might benefit from use of a palatal lift provided that conditions are such to justify prosthetic intervention. The palatal lift consists of a dental appliance with a posterior extension that lifts the soft palate so that it is the palate itself not the prosthesis that fills the gap (Peterson-Falzone et al., 2010). For the lift to work well there needs to be enough pharyngeal wall movement for contact with the edges of the raised palate (Golding-Kushner et al., 1995).

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Figure 4.6 Speech bulbs Reprinted with kind permission from Cleft Palate Speech, 2010. Copyright Elsevier

(b) Speech bulb Speech bulbs can be an effective permanent solution in patients with a serious degree of tissue deficiency where physical obturation of the VP valve is needed (Shprintzen, 1997). Such patients may present with no soft palate or with a significant anatomical defect, often following surgery. The appliance consists of a dental plate with a posterior extension with a bulb on it, which fills the space that cannot be closed by the soft palate and the pharyngeal walls during

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speech (Peterson-Falzone et al., 2010). During speech production of non-nasal consonants and vowels, the lateral pharyngeal walls move to make a seal around the bulb, whereas in quiet breathing, nasal airflow continues around the sides of the bulb (Sell et al., 2006). Construction of both palatal lift and speech bulb appliances requires a number of visits to the treatment centre. Videofluoroscopy and nasendoscopy procedures are usually employed during construction of the appliance, together with perceptual speech assessment. (c) Speech bulb reduction therapy/palatal training appliance For individuals with inconsistent VPD, Golding-Kushner et al. (1995) suggested ‘speech bulb reduction therapy’. This is gradual reduction in the size of a speech bulb, which the authors argue in some cases can be accompanied by increased VP motion, usually seen in lateral pharyngeal wall movement. This concept was first introduced by Blakeley in 1960 (Peterson-Falzone et al., 2006). However, speech bulb reduction programmes are not practised in the UK as they are considered to be experimental treatments and clinical indications remain in doubt (Sell et al., 2006). The ‘palatal training appliance’ consists of a U shape wire loop attached to the back of the dental plate, just touching the palate without applying pressure. The idea is that it would stimulate velar movement by sensory feedback. However, studies reporting effectiveness of this intervention have not produced sound scientific evidence supporting this principle (Peterson-Falzone et al., 2010; Sell & Grunwell, 2001).

Speech and language therapy/biofeedback for the management of VPD This can be an option in patients with inconsistent VPD who are able to achieve closure during speech (as shown in lateral videofluoroscopy and nasendoscopy), but who still present with evidence of VPD. This management option, including the evidence base, is explored further in Chapter 10.4.

Other non-surgical options Further options for the non-surgical management of VPD may involve a ‘watch and wait’ approach. This is relevant in cases where VPD is borderline and/or the child is very young. Inconclusive investigations due to lack of compliance for assessment procedures, or limited, delayed or disordered speech may create

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difficulties in establishing the nature, extent and impact of VPD on speech. Therefore, surgical management may best be deferred until there is certainty as to whether surgery is indicated and which surgical option is appropriate. Similarly, in borderline VPD cases, a conservative approach may be employed, especially in individuals who report no impact of borderline VPD on their communicative participation across different contexts (i.e., family, social, educational). A final management category involves individuals with speech diagnoses requiring onward referral, such as in cases of Developmental Verbal Dyspraxia (also known as Childhood Apraxia of Speech), severe hearing loss and various types and degrees of developmental disability. In some individuals with such diagnoses, VPD is manifested due to abnormal movement patterns of the soft palate, a VP activity that lacks rhythm and strength, or an impaired ability to monitor the nasal–non-nasal contrast in speech (Peterson-Falzone et al., 2010). In such individuals, referral to specialist centres may be the best option, as other types of management of their speech difficulties may be more appropriate than directly managing the VPD. However, it may be that addressing the VPD element of the speech difficulties is beneficial but decision making would need to take account of all contributory factors. In summary, there are a number of surgical and non-surgical treatment options for the management of VPD. Management decisions are always tailored to the individual and decision making may involve different members of the multidisciplinary team as well as the patient and family. Liaison with external agencies such as education professionals or other medical teams involved with the child may be also necessary.

• Case study: Helen Helen was born with a cleft of the hard and soft palate. There were no other anomalies present and no family history of cleft palate. Her initial palate repair took place at age 7 months but unfortunately her palate broke down at the central part of the repair, while the posterior part remained intact. This was re-repaired at the age of 14 months.

Assessment At Helen’s routine 18-month SLT assessment, hypernasality was noted, as well as backing of alveolar consonants and active nasal fricatives. Helen was

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offered input modelling sessions (see Chapter 7) and advice and activities were shown to parents on how to encourage sound production at home. Helen’s speech was reassessed at the age of 2:04 years. She presented with moderate hypernasality and inconsistent nasal emission accompanying sounds. Her fricative sounds /f v s z/ were substituted with active nasal fricatives, and her voiced plosives /b d/ were realised either as nasals /m n/ respectively, or inconsistently produced as the voiceless /p t/. There were inconsistent substitutions of consonants with /h/ in spontaneous speech. Helen’s speech presented with evidence of velopharyngeal dysfunction; however, her speech profile of active nasal fricatives, glottal articulation and inconsistent productions of plosives suggested articulation difficulties that had a further impact on intelligibility and perceptual assessment of palate function. Helen was offered speech therapy targeting the above speech errors and showed good progress with producing oral consonants with appropriate place and manner of articulation. Given the progress with consonant production, the surgeon and SLT jointly decided Helen would continue with speech therapy before attempting surgical intervention. This was to maximise her articulation potential (and velopharyngeal function potential) prior to reassessment of palate function to decide on type and extent of surgical intervention.

Investigation and management Investigation with lateral videofluoroscopy took place at the age of 3:09 years. This showed a short palate with good movement, a slightly large velopharyngeal space, posterior pharyngeal wall movement but a consistent large VP gap in all speech attempts. The surgeon decided Helen would benefit from a pharyngeal flap pharyngoplasty as she continued to present with moderate hypernasality and passive nasal emission accompanying consonants. She was by that stage able to produce all consonants appropriately but continued with speech therapy to generalise use of consonants in spontaneous speech. This case study illustrates that in some cases when VPD and nonoral articulation difficulties coexist, it may be appropriate for speech intervention to take place first before deciding on the type and extent of surgical intervention. Helen benefited from therapy to target non-oral cleft speech characteristics, as these had a significant impact on her speech intelligibility as well as perceptual assessment of palate function. Once oral

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articulation was established, instrumental and perceptual assessment revealed the extent of VPD and a surgical decision was based on this information. In Helen’s case, therapy was appropriate prior to surgery as she was able to achieve oral sounds and make progress with production of oral consonants with therapy alone. In other cases when VPD and articulation difficulties coexist but the child is unable to produce oral consonants, or sustain them in spontaneous speech, or speech therapy has not been successful in shifting non-oral and passive cleft speech characteristics, it may be appropriate for surgical intervention to take place first before articulation therapy.

• Case study: Michael

Michael was born with cerebral palsy. He was first seen in the Cleft Centre at the age of 9 years, following referral from his school SLT for hypernasal speech.

Assessment Michael’s speech profile consisted of moderate hypernasality, inconsistent nasal emission accompanying sounds, nasal realisations of consonants in spontaneous speech and weak consonants at single word level and careful speech. In addition, he presented with dysarthric speech characteristics such as slow/laboured speech, weak articulatory contacts and his intelligibility deteriorated in spontaneous speech. Investigation and management Michael was investigated with lateral videofluoroscopy and nasendoscopy. These revealed a long thick palate with minimal ability to move but there was some observable pharyngeal wall movement. This picture is consistent with a diagnosis of neurogenic VPD, which in Michael’s case was related to cerebral palsy. Surgical intervention was not considered appropriate in this case. Prosthetic intervention was therefore considered as an option to manage Michael’s VPD. As he presented with a long thick palate that would be able to make contact with the posterior pharyngeal wall if elevated, a palatal lift was considered appropriate. Michael was a cooperative boy who was able to tolerate having impressions taken for fabrication of the prosthesis. At the age of nearly 11 years his dental status allowed for retention of the appliance (which clips on to teeth) and he was able to show good

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understanding of what the appliance was for, why he needed to wear it and the importance of taking care of it. Furthermore, although Michael presented with dysarthric speech, his intelligibility improved with increased ability to build up intraoral pressure, as he achieved stronger consonants and sounded less hypernasal. This case illustrates that for significant VPD of neurogenic origin, surgical intervention may not be appropriate. Important considerations for successful management using prosthetic appliances are discussed earlier in the chapter, and Michael fitted the criteria. Finally, in cases of dysarthric patients such as Michael, it is important that careful assessment of all elements of speech production is undertaken and that improvement in speech intelligibility would be expected with wearing the prosthesis. In cases of severe dysarthria and limited articulation movements, prosthetic intervention may be of limited benefit.

References Bardach, J. (1995). Secondary surgery for velopharyngeal insufficiency. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach. St. Louis: Elsevier Mosby. Gardner, K. & Parr, G. (1996). Prosthetic rehabilitation of the cleft palate patient. Seminars in Orthodontics, 2(3), 215–219. Golding-Kushner, K., Cisneros, G. & LeBlanc, E. (1995). Speech bulbs. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach. St Louis: Elsevier Mosby. Goodacre, T. (2013). Personal communication. Oxford. Kuehn, D. & Moller, K. (2000). Speech and language issues in the cleft population: The state of the art. Cleft Palate-Craniofacial Journal, 37(4), 348–383. Kummer, A., Clark, S., Redle, E., Thomson, L. & Billmire, D. (2012). Current practice in assessing and reporting speech outcomes of cleft palate and velopharyngeal surgery: A survey of cleft palate/craniofacial professionals. Cleft Palate-Craniofacial Journal, 49(2), 146–152. Lipira, A., Grames, L., Molter, D., Govier, D., Kane, A. & Woo, A. (2011). Videofluoroscopic and nasendoscopic correlates of speech in velopharyngeal dysfunction. Cleft PalateCraniofacial Journal, 48(5), 550–560. Mazaheri, M. (1996). Prosthetic speech appliances for patients with cleft palate. In S. Berkowitz (Ed.), Cleft Lip and Palate: Perspectives in Management, Vol 2. San Diego: Singular Publishing Group.

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Mercer, N. & Pigott, R. (2001). Assessment and surgical management of velopharyngeal dysfunction. In A. Watson, D. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Muntz, H. (1992). Navigation of the nose with flexible fiberoptic endoscopy. Cleft PalateCraniofacial Journal, 29(6), 507–510. Ortichochea, M. (1983). A review of 236 cleft palate patients treated with dynamic muscle sphincter. Plastic and Reconstructive Surgery, 71(2), 180–186. Peterson-Falzone, S., Trost-Cardamone, J., Karnell, M. & Hardin-Jones, M. (2006). The Clinician’s Guide to Treating Cleft Palate Speech. St Louis: Elsevier Mosby. Peterson-Falzone, S., Hardin-Jones, M. & Karnell, M. (2010). Cleft Palate Speech, 4th Edn. St Louis: Elsevier Mosby. Riski, J. (2011). Secondary management and speech outcome. In S. Howard and A. Lohmander (Eds), Cleft Palate Speech: Assessment and Intervention. Chichester: Wiley-Blackwell. Sell, D. & Grunwell, P. (2001). Speech assessment and therapy. In A. Watson, D. Sell and P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Sell, D., Mars, M. & Worrell, E. (2006). Process and outcome study of multidisciplinary prosthetic treatment for velopharyngeal dysfunction. International Journal of Communication Disorders, 41(5), 495–511. Sell, D. & Pereira, V. (2011). Instrumentation in the analysis of the structure and function of the velopharyngeal mechanism. In S. Howard & A. Lohmander (Eds), Cleft Palate Speech: Assessment and Intervention. Chichester: Wiley-Blackwell. Shifman, A., Finkelstein, Y., Nachmani, A. & Ophir, D. (2000). Speech aid prosthesis for neurogenic velopharyngeal incompetence. The Journal of Prosthetic Dentistry, 83(1), 99–106. Shprintzen, R. (1995). Instrumental assessment of velopharyngeal valving. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach. St Louis: Elsevier Mosby. Shprintzen, R. (1997). Nasopharyngoscopy. In K. Bzoch (Ed.), Communicative Disorders Related to Cleft Lip and Palate, 4th Edn. Texas: Pro-Ed. Sommerlad, B. (2002). The management of cleft lip and palate. Current Paediatrics, 12, 43–50. Sweeney, T. (2011). Nasality: Assessment and intervention. In S. Howard & A. Lohmander (Eds), Cleft Palate Speech: Assessment and Intervention. Chichester: Wiley-Blackwell.

Bibliography Sell, D., Britton, L., Hayden, C., Phippen, G. & Russell, J. (2008). Speech and Language Therapy and Nasendoscopy for Patients with Velopharyngeal Dysfunction. RCSLT Position Paper. Royal College of Speech and Language Therapists. London.

5 Hearing Difficulties Lucy McAndrew

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Aim of this chapter This chapter examines: • What is otitis media with effusion? • Why hearing difficulties particularly occur in children with cleft palate or VPD. • The impact of hearing difficulties on speech and language development. • Indicators for concern. • Assessment and management of hearing difficulties.

Introduction Children with cleft palate or velopharyngeal dysfunction (VPD) often present with hearing difficulties. One of the primary causes of hearing loss in this

Figure 5.1 Coronal section through right ear showing middle ear

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group is the presence of otitis media with effusion, which has been reported to be almost universal in children with cleft palate (Szabo et al., 2010; Paradise et al., 1969). An additional hearing loss may also be present as part of an associated syndrome. The exact impact of otitis media on speech and language development remains uncertain (Chapman, 2004). However, the resultant hearing difficulties are likely to have consequences on speech and language development (Bamford & Saunders, 1990) and may also impact on well-being, socialisation, education and behaviour.

What is otitis media with effusion? Otitis media with effusion (OME) is more commonly referred to as ‘glue ear’. Rather confusingly, it is also known as middle ear effusion, chronic secretory otitis media or non-suppurative otitis media. In the middle ear (Figure 5.1), gases diffuse across the mucosal lining into the bloodstream causing the middle ear to be at constant negative pressure unless provided with a periodic supply of air via the Eustachian tube. In the presence of a well-functioning Eustachian tube, air enters into the middle ear from the nasopharynx through the Eustachian tube on swallowing or yawning. If tube function is impaired, for example, due to a cleft palate, swelling secondary to upper respiratory tract infection or enlarged adenoids, this can prevent adequate ventilation and clearing of mucus. This leads to increased negative pressure in the middle ear cavity and the production of fluid or ‘glue’ from the mucosal lining of the middle ear. This subsequently prevents the ear drum (tympanic membrane) from vibrating adequately. OME can present unilaterally or bilaterally and it is generally accepted as persistent glue ear if the presence of fluid has been confirmed for at least three months. OME is not usually infected but can be, and the effusion and any associated hearing loss often goes unnoticed by the child (Luthra et al., 2009).

Acute otitis media OME, sometimes referred to as chronic due to its long-term nature, should be distinguished from acute otitis media which is an isolated, usually selflimiting episode of inflammation of the middle ear. Acute otitis media often develops after an upper respiratory tract infection. The middle ear is usually

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infected and this is frequently accompanied by pain and fever in addition to hearing loss. The tympanic membrane can burst, releasing a discharge of pus, known as acute suppurative otitis media. Recurrent acute otitis media can be associated with OME and can occasionally lead to chronic suppurative otitis media, for example, in the case of a persistent tympanic membrane perforation. It has been reported that over 50% of children with cleft palate experience recurring episodes of acute otitis media with discharge (Van Cauwenberge et al., 1998).

Other predisposing factors for otitis media Aside from the presence of a cleft palate, there are a number of external influences on otitis media. First of all, both chronic and acute conditions are more common in winter. Nonbreastfed babies, a smoking environment and the use of dummies are all additional risk factors (Rovers et al., 2008; Hanafin & Griffiths, 2002).

What is the prevalence of otitis media with effusion in cleft palate? The association between cleft palate and hearing loss is well documented. It was first reported over a hundred years ago (Alt, 1878) and studies continue to report on the strong correlation between a cleft, OME and hearing difficulties (e.g., Kwan et al., 2011). Although OME may not be present at the neo-natal screen (Szabo et al., 2010), its onset is progressive and is most common in the first year of life (Li et al., 2007). Most children with cleft palate will develop OME and this often, but not always, results in a persistent conductive hearing loss, whereby sounds are unable to pass to the inner ear. To put this into context, up to one fifth of the general population experience otitis media at some point in their first five years of life.

What is the evidence? Viswanathan et al. (2008) reported an incidence of 82% hearing loss in babies with a cleft palate at age 2 months. The majority of this loss was found to be mild (86%), conductive (89%) and bilateral (84%). This was assessed objectively using auditory brainstem responses. Another study by Flynn et al. (2009) looked at the prevalence of OME

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in children with and without a cleft palate and found a significantly higher prevalence in the children with a cleft (75%) compared to those without a cleft (19%). This greater prevalence was significant at the ages of one, one and a half, three and five years old, illustrating the long-term nature of the effusion. They also reported that, of those with effusion, the degree of hearing loss was more significant in the cleft group than the non-cleft group. Thus, all children are at risk of developing OME and experiencing persisting hearing difficulties resulting from this, but those born with a cleft palate evidently have a substantially increased risk.

Syndromes In addition to a conductive hearing loss resulting from OME, a significant number of children with cleft palate will have a sensorineural hearing loss (SNHL) and/or a persistent conductive hearing loss (CHL) that is unrelated to middle ear effusion, as a consequence of an associated syndrome (Table 5.1). For example, ossicular malformations as seen in Oto-palato-digital syndrome can result in a CHL. Persistent CHL is also reported to be a feature of 22q 11.2 deletion syndrome. The cause of this is likely to be multi-factorial,

Table 5.1 Syndromes with hearing loss as a known feature Syndrome

Sensorineural Loss

Conductive Loss

Goldenhar or Hemifacial Microsomia





Stickler





Treacher Collins





22q 11.2 Deletion





Williams





Oto-palato-digital



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with immune deficiency as well as palatal and Eustachian tube dysfunction all being factors (Dyce et al., 2002). It has been suggested that up to 5% of children with a cleft palate will have an additional hearing difficulty that is unrelated to OME (Bardach et al., 1992). g Further Reading: Refer to end of chapter (reference section) for further reading on hearing difficulties in cleft palate with associated syndromes.

It is important to be aware of the possibility of additional hearing issues in children with both a cleft and a syndrome diagnosis as the loss may be mild and may be masked by the chronic hearing loss caused by OME (Chen et al., 2008).

Why do children with cleft palate get OME? Anatomical and functional differences have been reported in children with cleft palate. For example, a shorter Eustachian tube compared with age-matched children (Siegel et al., 1988) and differences in skull base factors related to the presence of a cleft (Carrie et al., 2000) have been found. Carrie et al. reported a significant difference in the sphenopalatine angles in the cleft palate group compared to the control group. Furthermore, their findings suggest that the greater the angle in the cleft palate group, the greater the protection against OME and associated hearing loss. Malpositioned palatal musculature such as abnormal insertion of the tensor veli palatini muscle and the course of the levator veli palatini muscle has also been reported (Arnold, 2005; Matsune et al., 1991). It has been proposed that these abnormalities cause obstruction of the Eustachian tube resulting in the increased incidence of OME. However, these findings remain controversial and continue to be debated. Children with a submucous cleft palate may also experience OME for the same reasons as a child with an overt cleft (Reiter et al., 2011).

The natural course of OME in children with cleft palate As the child grows, the Eustachian tube increases in size and the angle inclination becomes increasingly vertical, allowing more effective drainage of the middle

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ear. Consequently, spontaneous resolution of OME is likely to occur. Zheng et al. (2009) demonstrated an age-related decrease in the frequency of hearing impairment and abnormal middle ear function. In the majority of cases hearing will improve from age 6 years onwards (Møller, 1981).

Long-term consequences of OME/acute otitis media Both OME and acute otitis media can lead to tympanic membrane abnormalities. For example, the tympanic membrane can rupture with discharge of pus through the perforation. This can cause pain and antibiotic treatment is often required. In most cases the membrane heals spontaneously but occasionally repeated infection leads to a persistent perforation. This can result in thinning of the membrane (atrophy) or the development of scar tissue (tympanosclerosis). In some cases a persisting perforation which is non-symptomatic can in fact help by naturally ventilating the middle ear. Depending on the site of perforation, there may be a slight risk of cholesteatoma: a build up of dried skin (keratin) which, if left untreated, can lead to serious complications. Suspected cholesteatoma indicates the need for surgical exploration of the middle ear and the mastoid air cell system behind the middle ear.

The effect of palate surgery on Eustachian tube dysfunction Does surgery result in improved hearing? If abnormal anatomy can be repaired and function restored during palatal surgery (with or without grommet insertion) then one might speculate that a child’s hearing will be improved following primary repair. However, this is not always the case and again there is conflicting opinion in the literature. For example, Robinson et al. (1992) reported minimal improvement in middle ear status following palate repair in a cohort of 140 children. In contrast, some researchers stress the importance of surgical technique and suggest that a particular surgical approach will result in less otitis media both postoperatively and longer-term (Hassan & Askar 2007; Merrick et al., 2007) or the insertion of less sets of grommets (Smith et al., 2008). As palatal surgery aims to restore normal function it would seem likely that Eustachian tube function is also improved, resulting in better conductive hearing. However,

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surgical technique is likely to be influential in the outcome; the more accurate the restoration of the palatal muscles, the more likely Eustachian tube function will be normalised.

Care pathway How frequently is a child seen for audiology/ENT assessment? What happens? In view of the high incidence of OME and other hearing issues in children with cleft palate, it is important that access to regular audiological and ENT input is available from birth. The first assessment is the neonatal hearing screen routinely offered to all newborn babies in the UK. This takes place either in hospital or by community paediatric audiology shortly after birth. The screen involves presenting quiet clicking sounds into the ear called otoacoustic emissions (OAEs), which produce an echo from the cochlea. The OAEs can confirm normal inner ear (cochlear) function in the presence of normal middle ear function. Normal cochlear function cannot be confirmed, however, if the middle ear is filled with fluid. It is a ‘pass’ or ‘fail’ test indicating the need for further testing. Given the high incidence of middle ear effusion, it would seem likely that the young baby with a cleft would fail this screen. However, in practice many babies with a cleft pass the test (Szabo et al., 2010) and it has been suggested that this may be because the effusion has not yet developed so soon after birth. An absent echo indicates further assessment is required and an automated auditory brainstem response (ABR) is then performed locally. The ABR objectively assesses more frequency-specific hearing thresholds and can help identify any possible high-frequency SNHL. In a number of cleft centres, the coordinating ENT surgeon communicates with local ENT/audiology teams regarding the management of a child with a cleft and requests the results of the neonatal screen/ABR and regular monitoring as per local protocol. Due to discrepancies in local regional protocols, children born with a cleft may be followed up at differing intervals. The Clinical Standards Advisory Group (CSAG, 1998), which advised on standards of care for children and adults with cleft lip and/or palate, recommends that minimum audit records be taken for otoscopy and audiometry at pre-lip and pre-palate repair, three, five, ten, fifteen and twenty years of age. In light of the probable occurrence of otitis media at a young age, it would seem appropriate for children with cleft palate under three years to be assessed at a minimum of six-monthly intervals and then yearly until aged five.

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A: normal middle ear pressure; B: flat trace indicating little or no tympanic membrane movement, possibly due to fluid or grommet; and C: middle ear with negative pressure Figure 5.2 Schematic diagram of tympanogram

Investigations and hearing tests – what do the results tell us? Tympanometry Tympanometry measures tympanic membrane movement or rigidity (compliance) by delivering varied external air pressure to the tympanic membrane using a small probe. Maximal compliance is achieved when air pressure on either side of the tympanic membrane is equal. A graph of compliance versus air pressure is produced and the responses given suggest different conditions of the middle ear (Figure 5.2). For example, a peak at 0 decaPascals indicates a normal air-filled middle ear. A flatter peak reflects a stiff tympanic membrane as seen in ossicular fixation or, in the case of a child with a cleft, it may reflect a scarred membrane (tympanosclerosis) following repeated grommet insertion or perforation. A very high peak at 0 decaPascals can suggest a hypermobile or thin (atrophic) membrane. A peak to the left of the graph indicates negative pressure within the

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middle ear and a flat trace suggests either fluid, grommets in situ or a persistent perforated membrane. Tympanometry can be performed at any age and is usually routinely performed at a hearing test unless it is not possible or wise to do so such as in the case of excessive wax in the external auditory canal or a discharging infection.

Audiometry Audiometry refers to the testing of hearing ability at different frequencies and thresholds. The exact test performed will depend on the child’s age.

Figure 5.3 Example of an audiogram illustrating bone conduction within normal limits, a left-sided mild loss and a right-sided moderate loss

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Behavioural testing is not possible under six months of age due to lack of head control. From around seven months of age, head control is sufficient for distraction testing or visual reinforced audiometry (VRA), where the child turns towards a sound presented freefield and is rewarded with, for example, a puppet. At approximately age two and a half years, children ‘get wise’ to the puppet and turn in anticipation of a sound, and so performance testing is introduced, such as putting a figure on a bus in response to hearing a sound. For ear-specific information this test can be performed with headphones or inserts, if tolerated. From around four years of age, standard pure tone audiometry with headphones is possible, thus allowing a more informative assessment. Air conducted sounds can be presented either freefield or through headphones which provides a measure of overall hearing. If hearing difficulties are evident, further information can be obtained by testing bone conduction. This can prove challenging in the younger child as a bone conductor against their head may not be tolerated. The bone conductor sets the skull into vibration with pure tones, thereby mostly bypassing the middle ear route. Air conduction thresholds can be compared with bone conduction thresholds, giving information regarding the site of the hearing difficulty, more specifically whether it is a conductive or a SNHL. Responses are plotted on an audiogram (Figure 5.3), where 0dBs at each frequency represent the minimum level at which sound can be detected in normal hearing. In pure tone audiometry, from –10 to 20dBs is considered within the limits of normal hearing. A mild loss is considered to be around 20–40dBs, a moderate loss from 40–70dBs, a severe loss from 70–90dBs and a profound loss from 90dBs+. When both ears are being tested the overall loss is judged as the hearing level in the better ear. If there is an air–bone gap of greater than 10dBs, this would suggest a CHL. Glue ear usually results in a mild loss (Flynn et al., 2009) and never more than a moderate loss. Thus, if responses are greater than 60dBs, additional causes of hearing loss should be investigated. Where there is no air–bone gap and lower than normal thresholds, a SNHL loss is likely. It is important to be aware of the type of test used when interpreting test results, as higher thresholds would be expected on one test in comparison with another. For example, it is likely that higher, less subjective and therefore more accurate thresholds would be given on standard pure tone audiometry with head phones than on freefield performance testing.

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Impact on speech and language The evidence It is widely accepted that children with a cleft are at risk of speech and language difficulties. A number of studies have reported delays in receptive and expressive skills (Broen et al., 1998; Jocelyn et al., 1996; Scherer & D’Antonio, 1995). More recent studies have discussed the impact of a cleft on expressive abilities (Lu et al., 2010; Lohmander & Persson, 2008; Morris & Ozanne, 2003; Chapman et al., 2003; and see Chapman, 2008, for a review) and pragmatic deficits have also been highlighted (Frederickson et al., 2006). Additionally, literacy problems are reported to be a higher risk for children with a cleft (Chapman, 2011) although not all authors have found this to be the case (Collett et al., 2010). The impact of a cleft on speech and language outcomes continues to be clarified in the literature. A variety of aetiological factors is suggested including cognitive/linguistic immaturity (Chapman, 1993) or language/ phonological disorder (Eliason & Richman, 1990) in addition to structural or anatomical factors (McWilliams et al., 1990). The linguistic environment, including additional languages and the child’s cognitive level will also be factors to consider. Besides these potential variables, the high incidence of OME in children with a cleft is likely to impact on speech, language and communication skills although the degree the hearing loss contributes to any difficulties remains unclear.

• Research

In one study, the hearing, speech and language outcomes of 370 children with a cleft following palatal repair at 18–24 months were examined. The authors found that the group of children with CHL were significantly more severely affected on a variety of speech and language measures including phonology, morphology, syntax, vocabulary, auditory perception and language comprehension compared with the normal hearing group. The study concluded that hearing ability had a major influence on speech and language outcome (Schönweiler et al., 1999). Other researchers have reported a significant correlation between reduced hearing levels resulting from recurrent OME at 12–18 months and speech delay and low language outcomes at 3 years of age (Shriberg et al., 2000). Current evidence is sparse, however, indicating a need for larger-scale prospective

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studies of the impact of OME on speech and language development in children with a cleft.

Practical issues OME often (but not always) results in a mild (20–40dbs) and occasionally a moderate CHL. The loss is frequently variable with the consequence that a child may pass one hearing test and then fail the next and so forth. If the clinician is able to access an audiogram from a recent assessment (from parents or audiology department) this will provide helpful information regarding current degree and location of OME (left, right or bilateral) as well as the type of hearing loss. If parents report an infection with discharge then the child will have, or have had, a perforated tympanic membrane. The perforation can impact on hearing although the degree of loss will depend on the exact site and size of the perforation. The impact of a mild (to moderate) loss on speech and language development is not predictable. However, a bilateral 40dBs loss, for example, may result in one or more of the following phonetic/phonological or language consequences: Phonetic/phonological: • Difficulties with consonant discrimination (e.g., ‘sail’ vs. ‘tail’) • Omissions or difficulties with high frequency sounds such as ‘f,’ ‘s’ or ‘th’ • Absence of voiced/voiceless contrasts (e.g., ‘pin’ vs. ‘bin’) • Preference for anterior visible consonants • Weak syllable or final consonant deletion (e.g., ‘cu’ for ‘cup’) • Nasal realisations for vowel targets (leading to increased perception of hypernasality). Voice quality may also sound strained or even dysphonic. Language: • Delayed language comprehension • Difficulties with comprehension in quiet conversation • Difficulties with morphological endings (e.g., plural ‘s’, ‘Ben’s/Kim’s)

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• Simplified sentence structure (e.g., “daddy gone work”) • Use of more content than function words • More rigid, less flexible and stereotypical use of language. Table 5.2 summarises the indicators for concern in relation to parental report and observed behaviour/responses of the child. If a suspected hearing loss is thought to be impacting on speech, language or communication development and the child is not currently under audiology/ ENT management, a referral back to the local audiology team would be beneficial, following discussion and agreement from parents/carers.

Management of hearing difficulties Are ventilation tubes a good treatment option? A CHL associated with persistent OME can be managed with the insertion of ventilation tubes (usually grommets). These small tubes are inserted into the anterior inferior corner of the tympanic membrane under a general anaesthetic and work by keeping the middle ear ventilated and therefore free from further Table 5.2 Hearing loss – indicators for concern Indicators for concern for clinician Parental concern

Non-responsive behaviour

Lack of awareness/response to quiet voice

Not answering to name

Not following instructions

Misbehaviour

Reports of child being in a world of his own

Withdrawn/less socially responsive

Sits close to television

Delayed speech & language development

Requests increased television volume

History of hearing loss

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effusion. Used either unilaterally or more commonly bilaterally, the grommets, depending on the particular type used, remain in situ for approximately 12 months. Whilst in situ, normal hearing is usually restored. When the grommet extrudes, often spontaneously, it is sometimes found lying in the external auditory canal (or on the child’s pillow!) leaving the tympanic membrane to close behind naturally, or in some instances leaving a small hole at the site of the grommet. Unfortunately, the benefits to hearing are not always maintained following grommet extrusion as the glue ear frequently reoccurs and a further set may be recommended. Despite their benefits, grommets are not without risks. Some research suggests a higher incidence of tympanic membrane abnormalities such as tympanosclerosis and persistent perforations in children treated earlier and with more grommets compared to those who had no treatment or fewer insertions. Additionally, those who have undergone a greater number of grommet insertions are more likely to have a CHL (Phua et al., 2009; Goudy et al., 2006; Sheahan et al., 2002). One interpretation of such findings, however, is that these studies identify a sub-group of children who have more problems to begin with, resulting in more intervention. Indeed, a recent study stated that the perception that children managed with grommets experience higher complication rates is flawed and unfounded (Russell et al., 2012). Longer-lasting tubes can be used, for example, Goode T-tubes® or Permavent tubes. In theory, these should be effective for longer but due to their increased size they carry a greater risk of persistent perforations (Hawthorne & Parker, 1988) so are usually reserved for when standard grommets have not been effective. Persistent OME and consequential hearing loss in children with a cleft occurs during a critical period of development and acquisition of speech and language skills. Grommets can be a good treatment option during this time. However, due to an increased awareness of possible complications following grommet insertion, the routine use of grommet insertion at the time of primary palate repair is no longer universally supported (Phua et al., 2009; Ponduri et al., 2009). Instead, the use of selective and timely grommet insertion, when hearing is known to be consistently reduced, can optimise a child’s hearing at a crucial time in development and is now frequently advocated as the most effective way of managing OME (Merrick et al., 2007; Andrews et al., 2004; Sheahan et al., 2002). Wherever possible, grommet insertion should be coordinated with any other necessary surgical procedures to minimise the need for multiple anaesthetics (CSAG, 1998).

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Hearing aids An increasingly popular alternative management option for hearing impairment due to OME in children with a cleft is amplification using a hearing aid. A hearing aid is a non-invasive option which can be considered as a first line of management when OME has persisted or has reoccurred following grommet extrusion. The use of hearing aids in children with hearing loss can be a successful treatment option so long as the child does not suffer with recurrent suppurative otitis media. Its success, however, relies on the child’s ability to tolerate the behind the ear aid and close monitoring by the local audiology department to ensure accurate fitting, functioning and continued adjustments as required. For example, moulds may need to be replaced regularly due to growth or if damaged by otorrhoea. The child will also need parental/carer support and support from nursery or school to encourage and ensure effective compliance and use.

Grommets or hearing aid? The controversy as to whether grommets or a hearing aid is most beneficial is still under debate.

• Research

A survey sent to 594 consultant otolaryngologists in the UK regarding hearing aids versus grommets in the management of persistent OME found that 49.5% of respondents never or rarely recommended hearing aids (Ahmmed et al., 2001). However, the reasons given for reinserting grommets rather than offering an aid were not evidence-based, for example, that grommets are needed to stop middle ear complications or that parents preferred the surgical option. The possible complications of repeated grommet insertions are well documented and it is unlikely that parents are always given a balanced report on the treatment options in order to make an informed choice. The authors highlighted the need for a randomised control trial of hearing aids and ventilation tubes in the management of persistent and recurrent OME. In contrast, another researcher (Maheshwar et al., 2002) reported successful treatment of children with cleft palate using hearing aids as their first line of management. They reported that hearing loss caused by OME

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was satisfactorily treated and that grommets were advocated only in cases of non-compliance or recurrent suppurative otitis media. A low long-term middle ear complication rate was also reported. The choice of a hearing aid should be made on an individual basis. There are drawbacks to consider including possible stigma attached to wearing a hearing aid, with children being teased or bullied. The aid does not change the pathology and will not treat any discomfort or pain caused by an infection. It is only effective when it is being worn and it can be difficult to maintain at an appropriate level of amplification with a fluctuating loss. Nonetheless, a non-invasive approach where possible, with compliance and the appropriate support, would seem an advantageous option, particularly given the likely resolution of OME with advancing age.

Adenoidectomy Adenoidectomy is considered a treatment option for children with recurrent glue ear (Kadhim et al., 2007). However, in the presence of a cleft or VPD it is generally considered contraindicated as the procedure may result in increased hypernasality (Shprintzen & Bardach, 1995). Partial adenoidectomy has since been proposed to avoid post-operative VPD in patients with submucous cleft palate (Finkelstein et al., 2002) as well as in patients with an overt cleft (Tweedie et al., 2009). However, the literature presents conflicting opinions and some authors continue to advocate removal of the adenoids in the treatment of OME in children with a cleft palate followed by a pharyngoplasty if increased hypernasality occurs (Ruiter et al., 2009). Despite the lack of agreement, at the very least due to increased multidisciplinary working in cleft teams, the majority of ENT colleagues are aware of the speech implications of adenoidectomy in the cleft population.

How the clinician can impact on hearing difficulties Environmental manipulation As hearing loss associated with OME is often mild and fluctuating, it may not be necessary to manage with either grommets or a hearing aid, and a ‘watch and wait’ approach may be sufficient. It is therefore important for the clinician to be aware of any fluctuating loss and to refer back to audiology if there are indicators that hearing has deteriorated to a level which may require intervention (see indicators for concern, Table 5.2).

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In therapy, the clinician should ensure that the child can hear sufficiently for the task presented. It may be appropriate to postpone therapy until after grommets or a hearing aid have been fitted if, for example, targeting voiceless fricatives or plosives in word final position. It is advantageous to sit opposite rather than next to the child and the possibility that a child with a hearing loss may tire more easily should be factored in when planning therapy. With respect to advice to nursery or school, a child with a mild loss (or indeed any degree of loss) will benefit from sitting closer to the front of the class and being face to face with the teacher to maximise opportunities for lip reading, following facial expression, body language and gestures as well as for hearing. Keeping background noise to a minimum and attracting the child’s attention by using his name prior to giving an instruction will be helpful. Common comprehension strategies such as simplifying language, giving repetitions, rephrasing and checking comprehension by asking the child to repeat the instruction will all also benefit the child with a hearing loss.

Summary and conclusion Due to the high incidence of hearing difficulties in children born with a cleft palate, it is important for the clinician to be aware of a child’s current hearing status and to raise any suspected difficulties with parents/carers, referring on for assessment and management as necessary. This chapter has covered: • The nature and causes of possible hearing difficulties in children with VPD or cleft palate • Assessment procedures and interpretation of test findings • The impact of a hearing loss on speech and language development and indicators for concern • Management of hearing difficulties including practical advice for the clinician. The lack of agreement regarding best practice in managing children born with a cleft palate who present with OME shows that the debate continues and indicates the need for larger-scale randomised controlled trials. Again, although robust evidence is limited, there is general agreement that hearing loss can impact on speech and language development and so clinicians should remain vigilant to hearing problems and their potential impact on speech and language development.

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children with unilateral cleft lip and palate and two-stage palatal repair. Cleft PalateCraniofacial Journal, 45(1), 32–41. Lu, Z., Ma, L., Luo, Y. & Fletcher, P. (2010). The effects of unrepaired cleft palate on early language development in Chinese infants. Cleft Palate-Craniofacial Journal, 47(4), 400–404. Luthra, S., Singh, S., Nagarkar, A.N. & Mahajan, J.K. (2009). The role of audiological diagnostics in children with cleft lip & palate (CLP). International Journal of Pediatric Otorhinolaryngology, 73(10), 1365–1367. McWilliams, B.J., Morris, H.L. & Shelton, R.L. (1990). Cleft Palate Speech, 2nd Edn. Philadelphia: BC Decker. Maheshwar, A.A., Milling, M.A., Kumar, M., Clayton, M.I. & Thomas, A. (2002). Use of hearing aids in the management of children with cleft palate. International Journal of Pediatric Otorhinolaryngology, 66(1), 55–62. Matsune, S., Sando, I. & Takahashi, H. (1991). Insertion of the tensor veli palatini muscle into the eustachian tube cartilage in cleft palate cases. Annals of Otology, Rhinology & Laryngology, 100(6), 439–446. Merrick, G.D., Kunjur, J., Watts, R. & Markus, A.F. (2007). The effect of early insertion of grommets on the development of speech in children with cleft palates. British Journal of Oral & Maxillofacial Surgery, 45(7), 527–533. Møller, P. (1981). Hearing, middle ear pressure and otopathology in a cleft palate population. Acta Otolaryngolica, 92(5–6), 521–528. Morris, H. & Ozanne, A. (2003). Phonetic, phonological, and language skills of children with a cleft palate. Cleft Palate-Craniofacial Journal, 40(5), 460–470. Paradise, J.L., Bluestone, C.D. & Felder, H. (1969). The universality of otitis media in 50 infants with cleft palate. Pediatrics, 44(1), 35–42. Phua, Y.S., Salkeld, L.J. & de Chalain, T.M. (2009). Middle ear disease in children with cleft palate: Protocols for management. International Journal of Pediatric Otorhinolaryngology, 73(2), 307–313. Ponduri, S., Bradley, R., Ellis, P.E., Brookes, S.T., Sandy, J.R. & Ness, A.R. (2009). The management of otitis media with early routine insertion of grommets in children with cleft palate—a systematic review. Cleft Palate-Craniofacial Journal, 46(1), 30–38. Reiter, R., Brosch, S., Wefel, H., Schlömer, G. & Haase, S. (2011). 1.The submucous cleft palate: Diagnosis and therapy. International Journal of Pediatric Otorhinolaryngology, 75(1), 85–88. Robinson, P.J., Lodge, S., Jones, B.M., Walker, C.C. & Grant, H.R. (1992). The effect of palate repair on otitis media with effusion. Plastic & Reconstructive Surgery, 89(4), 640–645. Rovers, M.M., Numans, M.E., Langenbach, E., Grobbee, D.E., Verheij, T.J. & Schilder, A.G. (2008). Is pacifier use a risk factor for acute otitis media? A dynamic cohort study. Family Practice, 25(4), 233–236. Ruiter, J.S., Korsten-Meijer, A.G. & Goorhuis-Brouwer, S.M. (2009). Communicative

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abilities in toddlers and in early school age children with cleft palate. International Journal of Pediatric Otorhinolaryngology, 73(5), 693–698. Russell, C., Black, O., Dutt, D., Ray, A., Devlin, M. & Wynne, D. (2012). Are ventilation tubes (grommets) in cleft children truly associated with increased complication rates? Results of a nested case control study of cleft and non-cleft children. British Journal of Oral and Maxillofacial Surgery, 50 (Supplement 1), S2–S3. Scherer, N.J. & D’Antonio, L.L. (1995). Parent questionnaire for screening early language development in children with cleft palate. Cleft Palate-Craniofacial Journal, 32(1), 7–13. Schönweiler, R., Lisson, J.A., Schönweiler, B., Eckardt, A., Ptok, M., Tränkmann, J. & Hausamen, J.E. (1999). A retrospective study of hearing, speech and language function in children with clefts following palatoplasty and veloplasty procedures at 18–24 months of age. International Journal of Pediatric Otorhinolaryngology, 50(3), 205–217. Sheahan, P., Blayney, A.W., Sheahan, J.N. & Earley, M.J. (2002). Sequelae of otitis media with effusion among children with cleft lip and/or cleft palate. Clinical Otolaryngology & Allied Sciences, 27(6), 494–500. Shprintzen, R.J. & Bardach, J. (Eds) (1995). Cleft Palate Speech Management: A Multidisciplinary Approach (p.259). St. Louis: Elsevier Mosby. Shriberg, L.D., Friel-Patti, S., Flipsen, P. Jr. & Brown, R.L. (2000). Otitis media, fluctuant hearing loss, and speech-language outcomes: A preliminary structural equation model. Journal of Speech, Language and Hearing Research, 43(1), 100–120. Siegel, M.I., Sadler Kimes, D. & Todhunter, S.S. (1988). Eustachian tube cartilage shape as a factor in epidemiology of otitis media. Proceedings of the 4th International Symposium. In D.J. Lim, C.D. Bluestone, J.O. Klein & J.D. Nelson (Eds), Recent Advances in Otitis Media (pp.114–117). Burlington, Ontario: BC Decker. Smith, L.K., Gubbels, S.P., MacArthur, C.J. & Milczuk, H.A. (2008). The effect of the palatoplasty method on the frequency of ear tube placement. Archives of Otolaryngology – Head & Neck Surgery, 134(10), 1085–1089. Szabo, C., Langevin, K., Schoem, S. & Mabry, K. (2010). Treatment of persistent middle ear effusion in cleft palate patients. International Journal of Pediatric Otorhinolaryngology, 74(8), 874–877. Tweedie, D.J., Skilbeck, C.J., Wyatt, M.E. & Cochrane, L.A. (2009). Partial adenoidectomy by suction diathermy in children with cleft palate, to avoid velopharyngeal insufficiency. International Journal of Pediatric Otorhinolaryngology, 73(11), 1594–1597. Van Cauwenberge, P.B., De Moor, S.E.G. & Dhooge, I. (1998). Acute suppurative otitis media. In H. Ludman and T. Wright (Eds), Diseases of the Ear, 6th Edn. London: Arnold. Viswanathan, N., Vidler, M. & Richard, B. (2008). Hearing thresholds in newborns with a cleft palate assessed by auditory brain stem response. Cleft Palate-Craniofacial Journal, 45(2), 187–192. Zheng, W., Smith, J.D., Shi, B., Li, Y., Wang, Y., Li, S., Meng, Z. & Zheng, Q. (2009). The natural history of audiologic and tympanometric findings in patients with an unrepaired cleft palate. Cleft Palate-Craniofacial Journal, 46(1), 24–29.

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Further Reading Barozzi, S., Soi, D., Comiotto, E., Borghi, A., Gavioli, C., Spreafico, E. et al. (2012). Audiological findings in Williams syndrome: A study of 69 patients. American Journal of Medical Genetics, Part A, 158A(4), 759–771. Marres, H.A. (2002). Hearing loss in the Treacher-Collins syndrome. Advances in OtoRhino-Laryngology, 61, 209–215. Skarzynski, H., Porowski, M. & Podskarbi-Fayette, R. (2009). Treatment of otological features of the oculoauriculovertebral dysplasia (Goldenhar syndrome). International Journal of Pediatric Otorhinolaryngology, 73(7), 915–921. Szymko-Bennett, Y.M., Mastroianni, M.A., Shotland, L.I., Davis, J., Ondrey, F.G., Balog, J. et al. (2001). Auditory dysfunction in Stickler syndrome. Archives of Otolaryngology – Head & Neck Surgery, 127(9), 1061–1068. Zarchi, O., Attias, J., Raveh, E., Basel-Vanagaite, L., Saporta, L. & Gothelf, D. (2011). A comparative study of hearing loss in two microdeletion syndromes: Velocardiofacial (22q11.2 deletion) and Williams (7q11.23 deletion) syndromes. Journal of Pediatrics, 158(2), 301–306. Zaytoun, G.M., Harboyan, G. & Kabalan, W. (2002). The oto-palato-digital syndrome: Variable clinical expressions. Otolaryngology-Head and Neck Surgery, 126(2), 129–140.

6 Voice Issues Leda Koutsoulieri

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Aim of this chapter This chapter discusses the relationship of cleft lip and palate/ velopharyngeal dysfunction (VPD) and voice. It will briefly explore the complex issues surrounding assessment and management of dysphonia and VPD when the two coexist. It is written from the perspective of a specialist in cleft/VPD but, as will become clear, management requires close collaboration between cleft/VPD clinicians and voice clinicians.

A brief review of the relationship between voice and resonance The human voice fulfils many roles in the process of human communication; it makes speech and language audible, has both a paralinguistic and a linguistic role and serves as a powerful conveyor of identity and emotional state. Overall, the voice contributes to the way in which people relate to each other. It is because of these varied functions that voice impairment or loss can be very distressing to the individual (Mathieson, 2001).

Figure 6.1 Supraglottal tract

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Voice and speech are so interlinked that they are often understood to be synonymous by the general public. Speech production involves a number of organs, including the lungs, trachea, larynx and vocal tract and is initiated with air from the lungs moving up the trachea and into the larynx. There, it passes between the vocal folds which vibrate to produce voice. The sound then passes through the pharynx, oral and nasal cavities and is shaped by the articulators to form speech sounds (Ladefoged, 1993).

What is voice? Voice is generated by the rapid opening and closing of the vocal folds; this vocal fold vibration produces an acoustic signal, or note, which we call voice (O’Connor, 1973). Voice is created in the larynx but is filtered by the supraglottal tract, i.e., it is shaped by the pharynx, the oral and the nasal cavities. Therefore, the voice heard by the listener is the acoustic product of the entire vocal tract, not just the sound produced by the larynx alone (Mathieson, 2001).

• Terminology

Supraglottal tract: the vocal tract above the level of the larynx (see Figure 6.1).

What is resonance? Resonance has been defined as “the vibratory response of a body or air-filled cavity to a frequency imposed on it” Wood (1971), in Peterson-Falzone et al. (2010), p.221. Speech resonance is the result of the transfer and modification of the acoustic signal produced in the larynx through the vocal tract. An acceptable resonance is one that is perceived to have a balance of oral and nasal resonance; for English vowels and vocalic consonants it is predominantly oral, with some contribution of nasal resonance (Peterson-Falzone et al., 2006). The above definitions make it clear why voice and resonance are so interlinked, even though they describe different things. Consequently, when assessing voice quality and resonance – such as in individuals with cleft palate and/or VPD – one should consider both glottal (laryngeal) and supraglottal (above larynx) features as they form part of the whole system.

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Voice and resonance disorders Disorders at the level of the larynx and those occurring above it are classified differently. Voice disorders occur at the laryngeal level and are also referred to as disorders of phonation. Resonance disorders occur above the level of the larynx, and refer to the deviations in balance of oral and nasal resonance. These are associated with supralaryngeal physical or functional problems, most commonly velopharyngeal structure and function (Henningsson et al., 2008; Wyatt et al., 1996).

• Note

See Chapter 3.2 for definitions of resonance disorders. It is perhaps this distinction of laryngeal versus supralaryngeal levels that has paved the way for clinicians to focus on either one or the other, and has seen the clinical specialities of voice disorders and velopharyngeal function disorders develop separately within speech and language therapy. Boone (2004) describes the emergence of the two specialities in the 1950s, with different approaches to remediation of velopharyngeal problems in defects like cleft palate versus remediation of voice problems secondary to organic pathologies or laryngectomy. In current UK clinical practice, speech and language therapists generally specialise in assessment and management of either voice disorders or cleft palate/VPD speech disorders.

Prevalence of voice disorders It is difficult to define normal voice because each individual voice has its own distinct features. Furthermore, the voice of the same person also varies, showing different vocal features depending on mood, illness, tiredness and communication context (Mathieson, 2001). It can also be challenging to describe a disordered voice; a voice disorder has been defined as a deviation in quality, pitch, loudness or flexibility when compared to the voices of others of similar age, gender and cultural group (Aronson, 1990). Voice disorders range from complete voice absence to varying degrees of vocal impairment. They are the manifestation of disordered laryngeal, respiratory and vocal tract function and may reflect among others, structural, neurological, psychological and behavioural problems (Mathieson, 2001). There is limited information on the prevalence of voice disorders in the general paediatric population, due to differences in assessment

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methods, populations studied and voice criteria. In a recent large prospective epidemiological study of eight-year-old UK children, Carding et al. (2006) examined the prevalence of childhood dysphonia. They quoted an abnormal/ atypical voice quality prevalence of 6% when assessed by a clinician and 11% when reported by a parent.

Voice disorders in cleft/VPD The prevalence of voice disorders in the cleft palate/VPD population is also not well established in the literature. There are reports quoting figures between 17% and 41% of children and adults with dysphonia, with a decline in prevalence towards puberty (Cavalli, 2011). It has been suggested that the occurrence of voice disorders in individuals with cleft palate is not well understood because it varies depending on velopharyngeal adequacy and the criteria used for defining the voice problem (Peterson-Falzone et al., 2010). In addition to the lack of clear prevalence data, there is also debate in the literature as to whether individuals with cleft palate/VPD are indeed more at risk of developing a voice problem. However, there is evidence that patients with cleft palate and a syndromic diagnosis are at greater risk, as outlined below (Cavalli, 2011; Witzel, 1995): • Apert syndrome: abnormal pitch and calcification of the larynx • Hemifacial Microsomia: Diplophonia • Ectrodactyly-Ectodermal Dysplasia-Clefting Syndrome: breathiness secondary to poor lubrication of the vocal folds •  22q11.2 deletion syndrome: vocal fold paralysis and glottic web.

Nature and causes of voice disorders in cleft/VDP In the general paediatric population, vocal fold nodules and inflammation are among the most common causes of dysphonia (De Bodt et al., 1995). Common risk factors for paediatric chronic dysphonia have been reported to include childhood hearing problems, history of shouting, frequent upper respiratory tract infections, childhood asthma, history of allergies, large family (sibling) dynamics (Mathieson, 2001). A recent study suggested that gender (male children were more susceptible than female) as well as number of older siblings in the household were the most significant risk factors for developing dysphonia in children (Carding et al., 2006). Regarding the nature of voice disorders in cleft/VPD, there is consensus in

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the literature that the majority of children that present with dysphonia have a muscle tension voice disorder, with or without reactive mucosal changes (i.e., vocal fold nodules, diffuse inflammation/oedema) (Cavalli, 2011). Various studies have provided evidence of laryngeal findings in individuals with cleft palate (in Van Lierde et al., 2004), including: • Chronic inflammation • Hyperplasia of vocal folds • Vocal fold nodules • Oedema. Vocal quality characteristics in individuals with cleft palate as described in the literature (Van Lierde et al., 2004), include:

• Breathiness, hoarseness • Fundamental frequency differences • Soft voice syndrome, aspirate phonation • Vocal pitch differences, limited voice pitch range • Reduced or excessive loudness. However, when describing the nature of dysphonia in children with cleft/ VPD, contradictory evidence has also been reported. Some studies describe participants’ voices as soft or aspirated and others as strangled with excessive tension. Some report normal fundamental frequency (F0) while some others report a lower F0 in females, higher in males, or excessively high or low pitch. A consensus has not been reached as to the nature of voice disorders in this population, which most probably reflects the variability of the symptoms and the individuals studied as well as the methods used to study these disorders. Regarding the causes of dysphonia in children with cleft/VPD, various reasons have been described. It has been hypothesised that the speaker uses excessive laryngeal valve action and laryngeal constriction to compensate for inadequate velopharyngeal closure (Peterson-Falzone et al., 2006; Grunwell & Sell, 2001). Conversely, soft voice syndrome, which has been described in the cleft palate population, is a compensatory strategy where the speaker may intentionally talk more softly to minimise/disguise hypernasality and nasal emission (Peterson-Falzone, 2006). Vocal fold pathology may also develop in children who consistently use glottal articulation, with increased laryngeal hyperfunction (Witzel, 1995). Other explanations for dysphonia in

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the cleft palate/VPD population have been attributed to tongue retraction and rigidity, persisting learned compensations even after surgical correction, and psychological factors (Cavalli & Pinkstone, 2005). Kuehn and Moller (2000) argue that although there is no evidence suggesting that laryngeal or phonatory structures of individuals with clefts are different to the non-cleft population, the aetiology of dysphonia in the former is best explained by the interaction of velopharyngeal dysfunction and laryngeal compensatory behaviour. Finally, it should not be assumed that dysphonia in children with cleft/VPD is necessarily related to this condition. General health can be a primary factor in paediatric vocal abuse, or contribute to the problem. Contributing factors may include frequent upper respiratory tract infections, hearing loss, allergic reactions and asthma (Mathieson, 2001).

Voice and VPD: What is the evidence? There is a low volume of studies dealing with voice issues in individuals with cleft palate/VPD. In terms of dysphonia in the presence of VPD, below are some reports that have examined this link.

• Research

Guyette et al. (2000) studied the effect of VPD on aerodynamic measures of laryngeal function in children with cleft palate. They compared children with complete velopharyngeal (VP) closure versus incomplete closure. Transglottal pressure, transglottal airflow and laryngeal resistance were measured. Interestingly, the authors found that children in the complete VP closure group showed significantly higher laryngeal resistance and transglottal pressure (suggesting increased effort when speaking) than subjects with incomplete closure. The authors hypothesised that there may be a subgroup of children that achieve complete VP closure in speech only by using excessive muscular effort which then leads to excessive vocal effort, compared to the incomplete VP closure group. D’Antonio et al. (1988) looked into the prevalence of laryngeal/voice findings in children and adults with both cleft and non-cleft VPD. The participants received speech assessment, otolaryngologic evaluation, endoscopy of VP mechanism and the larynx (the latter when there were concerns about voice/laryngeal function). Recordings of nasal airflow and

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oral air pressure were made, as well as estimates of subglottal air pressure. Results showed that 41% of participants showed either laryngeal findings, or abnormal voice findings or both (of those 21% had vocal fold nodules or thickened vocal folds). Hoarseness was the most common symptom followed by abnormal pitch and reduced loudness. Despite the high prevalence of those findings, interestingly, results showed no significant relationship between endoscopic/aerodynamic findings of VPD and presence of laryngeal/voice findings. The authors concluded that patients with VPD should receive perceptual and structural assessment that includes laryngeal function, given the high percentage of laryngeal/voice findings in their study. However, they also suggested that the link between VPD and laryngeal/voice abnormalities is not clear, as their data failed to establish a clear relationship between the two. Findings to support this suggestion also came from Hamming et al. (2009). In their study of children with cleft palate, the authors reported a hoarseness rate of 20% in ages 3–4 years and 18% in ages 6–7 years, which they argue is similar to that of the non-cleft population. Most interestingly, they did not find a relationship between severity of VPD and hoarseness and reported a similar rate of hoarseness when patients had VPD and when they did not. Leder and Lerman (1985) analysed spectrograms of adults with cleft palate presenting with mild hypernasality and severe hypernasality, and adults without cleft. There is little published data on vocal characteristics of adults with cleft palate as there is often remission of voice problems in adulthood. However, results from this study showed that in speakers with severe hypernasality, their vocal folds were inappropriately adducted to provide a constriction below the level of the inadequately functioning VP port to minimise the nasal loss of air. The authors support the hypothesis that a cause of voice disorders in individuals with cleft palate/VPD is due to compensatory laryngeal valving activity, secondary only to significant hypernasality. Van Lierde et al. (2004) studied the vocal quality characteristics of children with cleft palate and compared these findings with normative data. Children with cleft palate and mild hypernasality were evaluated using both subjective (videolaryngostroboscopy and auditory perceptual evaluation) and objective (aerodynamic, voice range, acoustic and Dysphonia Severity Index (DSI)) measures. Their results showed that a laryngeal pathology

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was observed in 32% of the children. Bilateral vocal nodules and muscle tension dysphonia were the most frequently observed laryngeal/voice findings. Furthermore, all their participants with cleft palate demonstrated significantly lower DSI value compared to the norm (showing more impaired vocal quality). However, in terms of their overall voice profile (as evaluated by both the subjective and objective measures) only the male children showed a vocal quality significantly lower than the norm, with presence of hoarseness and roughness. The authors therefore argued that genderrelated differences exist in voices of children with cleft palate and that boys are more vulnerable to dysphonia, a finding that has also been reported in the non-cleft paediatric population. Hocevar-Boltezar et al. (2006) described a dysphonia prevalence of 12.5% in children with cleft palate, which they argued is similar to prevalence reported in children without cleft. Furthermore, all participants had a complete ENT evaluation, including a hearing test, revealing that 56% had otitis media with effusion, with half of them requiring surgical intervention. The authors argued that long-lasting hearing loss may be an important factor, possibly influencing the prevalence of dysphonia in children with cleft palate, as hearing-impaired children often speak loudly and may abuse their voices. Also, further ENT pathology found in this study (impaired nasal breathing due to deviated septum and rhinitis) may also play a role in voice symptoms as it adds to vocal vulnerability through the drying of laryngeal secretions (Cavalli, 2011). In summary, some studies suggest that there is an increased risk of voice pathology in individuals with cleft/VPD compared to the general population, although others report a similar prevalence. Furthermore, the relationship between voice and VPD is not clear, as some studies report a link between mild VPD and voice pathology, whilst others link only severe VPD with voice symptoms. It has been argued that learned hyperfunctional laryngeal gestures (i.e., false fold and vocal fold hyperconstriction and supraglottic narrowing) may persist even in cases where the VPD is resolved (Cavalli, 2011).

• Note

It is important to remember that it is difficult to make direct comparisons between studies, due to methodological differences, particularly concerning

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assessment methods and populations studied, as the latter can widely differ between investigations.

Voice assessment In the UK there are three main voice quality assessment measures in use: the GRBAS scale (most commonly used); the Vocal Profile Analysis (VPA) scheme; and the Buffalo III Voice Profile (Carding et al., 2000). These tools can be used interchangeably, depending on the purpose of the task and the experience of the user. The GRBAS scale, which has been recommended as the minimum standard of vocal perceptual evaluation in the UK, evaluates hoarseness on five scales: Grade (degree of abnormality), Roughness, Breathiness, Asthenia (weakness) and Strain. The Vocal Profile Analysis (VPA) Scheme provides a list of items demonstrating a variety of vocal qualities and phonetic gestures. It allows the listener to both describe and analyse voice quality as it profiles individual vocal characteristics. It requires regular listening skills practice and a two-day training course before it can be used. The Buffalo III Voice Profile allows for rating of 12 aspects of voice production, such as pitch, loudness, breath supply, rate, muscles, laryngeal tone, speech anxiety, intelligibility, voice abuse, oral and nasal resonance and overall voice rating. Decisions on the most suitable scheme to use depend on factors such as the purpose of assessment (e.g., clinical practice, research), the level of detail required to direct treatment and the experience of the user, as some assessments need specialised training before use (Mathieson, 2001; Carding et al., 2000).

How do we assess voice in the cleft/VPD population? The importance of assessing voice alongside palate function and cleft speech characteristics (CSCs) has been suggested as best practice. However, in clinical practice, abnormalities in the anatomy and function of the larynx have not been well documented in patients with cleft palate/VPD, as it is often not part of the routine assessment protocol. Where appropriate, it has been suggested that the entire vocal tract, including the larynx, is visually assessed when problems with either resonance or laryngeal phonation are suspected (Witzel, 1995). D’Antonio and Scherer (1995) also advocate the assessment of the entire vocal tract, as the velopharynx is only one part of it; however, they note that:

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“... there is great pressure in some settings for the speech pathologist to focus primarily or even exclusively on resonance and VP function with respect to surgical success and further management” D’Antonio & Scherer (1995), p.177. Assessment of speech for individuals with cleft palate/VPD speech in the UK uses the Great Ormond Street Speech Assessment (GOS.SP.ASS) (Sell et al., 1999). (See Resource CD.) This includes assessment of: • Resonance • Nasal emission and nasal turbulence • Grimace • Consonant production and cleft speech characteristics (CSCs). Other areas are also observed and recorded including visual appearance of speech, oral examination and voice. In describing voice, the descriptors used are: normal voice; dysphonic voice; reduced volume. In their original paper, the authors state that “...any unusual voice quality should be categorised as dysphonic” Sell, Harding & Grunwell (1999), p.24. There is another assessment tool that has been developed specifically for use in inter-centre audit studies of cleft speech to systematically record and report speech outcomes; the Cleft Audit Protocol for Speech – Augmented (CAPS-A) (John et al., 2006). This includes assessment of the following parameters: • Intelligibility • Voice quality • Resonance • Nasal airflow • Grimace • Cleft speech characteristics (CSCs). Voice quality is documented using a binary system where voice is described

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as either ‘normal’ or ‘distinctive/abnormal’. Both these assessments therefore recognise the importance of assessing voice and include it as a parameter that needs clinical evaluation in individuals with cleft/VPD.

Assessment issues As mentioned earlier in this chapter, voice and resonance are interlinked so there is some overlap in the perceptual assessments used for both voice and VPD parameters. However, the cleft/VPD speech assessments include only a limited evaluation of voice. Similarly, the voice assessments do not all contain measures of resonance, for example, the GRBAS scale does not contain supraglottic parameters. Similarly, even though the VPA does include such parameters, the velopharyngeal section uses different descriptive terms and scale to the cleft speech assessments, which makes comparisons difficult. The Buffalo III Voice Profile includes analysis of nasal and oral resonance, again using a different system (Carding et al., 2000). There is currently no formal assessment focusing on both voice and velopharyngeal function. Therefore, when assessing a child with cleft palate/VPD it is important to recognise and record the presence of dysphonia and, if persisting, to refer to a voice specialist for further evaluation of voice. A protocol for voice assessment in individuals with cleft palate has been described by McWilliams et al. (1990), where the first step is to carefully listen to voice characteristics and determine whether there is evidence of dysphonia. If such evidence exists, a thorough perceptual voice evaluation should take place, including a case history to determine whether the symptoms are recent or chronic, typical for the child or atypical and related to a cause such as a cold or recent vocal abuse. It has been suggested that the presence of excessive loudness, reduced loudness or abnormal use of pitch range are often behavioural variables that are not usually signs of vocal fold pathology. However, features such as harshness, breathiness or phonation breaks may be indicative of pathology such as vocal fold nodules, thickening and oedema (D’Antonio and Scherer, 1995). Patients with such symptoms should be referred to an ENT clinic for evaluation. An instrumental assessment may be necessary, including laryngeal examination and vocal tract imaging (i.e., laryngoscopy, stroboscopy, X-ray imaging), physiological assessment (e.g., electrolaryngography) and acoustic analysis (sound spectrography, phonetogram), among others (Cavalli & Pinkstone, 2005).

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g Further Reading: For further information on instrumental and perceptual evaluation of voice, see Chapter 13 in: Mathieson, L. (2001). Greene and Mathieson’s The Voice and its Disorders, 6th Edn. London: Whurr.

Why is voice assessment important for individuals with cleft palate/VPD? Judgements about resonance are difficult to make in the presence of voice quality deviations as these can mask true hypernasality (Kuehn & Moller, 2000). Assessment of resonance is based on judgements of vocal resonance so, if a child presents with dysphonia, assessment of nasal resonance is more difficult (Wyatt et al., 1996). Similarly, VPD can affect evaluation of voice. It has been reported that marked hypernasality can mask dysphonia, and in some cases a voice problem may not be identified until treatment for VPD is under way. This is because hypernasality affects the voice signal in a number of ways, i.e., by shifting the first formant, providing extra resonances and anti-resonances and noise between the formants, among others (Cavalli, 2011). It is clear then that voice evaluation in cleft palate patients is best practice not only to detect the presence of voice deviations and laryngeal pathology but also for accurate assessment and management of VPD.

• Research

Imatomi (2005) suggested that hypernasality ratings tend to be difficult or unstable in the presence of voice deviations and described a study that investigated the effects of breathy voice on ratings of hypernasality, using synthesised speech. In this study, speech samples of varying degrees of hypernasality and breathiness were rated by speech and language therapists. The author concluded that the presence of breathiness raised mild hypernasality while it reduced severe hypernasality. Therefore, it was argued that caution should be applied in assessment of cleft patients with dysphonia, as resonance ratings can be directly affected.

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Intervention for voice disorders Voice disorders can be treated with voice therapy, medication, surgery or a combination of the above, depending on the aetiology. The majority of voice disorders can be treated with voice therapy; this type of therapy uses behavioural strategies to change vocal behaviours in order to minimise or resolve the vocal disorder. Phonosurgery (surgery to improve phonation) aims to improve laryngeal structure, and potentially function, by removing abnormal laryngeal tissue unresponsive to voice therapy. However, even in cases of successful surgery, voice therapy can be part of the rehabilitation process. Medication may also be prescribed as part of the treatment process (Mathieson, 2001). Voice therapy is the treatment of choice for most cases of voice disorders arising from muscle tension dysphonia and vocal fold nodules. Given that most children with non-syndromic cleft palate/VPD fall into this category, voice therapy is appropriate to consider in the child’s overall speech and language therapy management (Cavalli, 2011). When nodules do not improve with therapy, surgery may be necessary. It has been suggested that surgery is only recommended as a first treatment option, when nodules are very big, the voice is very rough, and a fast improvement is required. Surgery is rarely indicated in children because, during development, nodules can be re-absorbed into the vocal folds and in the absence of vocal behaviour management, relapses are possible (Trani et al., 2007). Describing voice therapy techniques is beyond the scope of this chapter; however, the general principles of voice therapy are described below as these also apply to children with cleft/VPD in need of voice therapy.

Goals of voice therapy Mathieson (2001) describes the general aims of voice therapy as follows: • Restore normal voice • Improve vocal profile • Improve/restore laryngeal function • Protect the patient from regressing or developing further vocal problems • Eliminate vocal tract discomfort associated with phonation • Enable the patient to reach vocal potential.

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Treatment goal setting should take into consideration the following: • Laryngoscopic findings • Instrumental and perceptual acoustic profile • Vocal tract discomfort and phonatory effort • Emotional status • Non-phonatory vocal tract behaviours (e.g., throat clearing). It is important to remember that although normal voice may be an appropriate target for some cases of organic-based paediatric voice disorders, for others, e.g., vocal fold palsy or laryngeal web, the goal will be to maximise the communication potential of the patient (Cavalli, 2011).

Indirect/direct approaches When thinking about offering voice therapy in young children, it is important to first consider whether they are developmentally ready for this type of therapy. Voice as a concept is relatively abstract and some children may not be able to recognise and understand what it is they are trying to achieve or change. For younger children it may be more appropriate to use an indirect approach and work through significant others, adapting conversational rules according to the child’s level and needs, as well as adapting the environment. Advice often given to families is to: • Respect conversational shifts • Limit background noise during talking • Respect vocal advice • Allow the child to play and rest his/her voice • Allow time to relax and listen • Avoid excessive heating and air-conditioning • Keep the mucous membranes hydrated. (Trani et al., 2007) The use of augmentative/alternative devices (e.g., voice amplifiers) as well as counselling and support, could also be employed (Cavalli, 2011). These

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interventions are even more relevant in cases of coexisting voice disorders and VPD, where the impact on speech may be even greater. Direct therapy approaches aim to change the vocal physiology, and include muscle relaxation, breathing exercises, production of easy onset voice, biofeedback training and manual laryngeal musculoskeletal massage (Lee & Son, 2005). Young children may not be responsive to this approach, so patient selection is important when offering this type of input. Direct voice therapy treatment programmes have generally been described as having three stages, as outlined below (Cavalli, 2011; Lee & Son, 2005): Stage 1: Developing awareness of the problem, voice production and laryngeal tension. Stage 2: Developing vocal control and targeted vocal behaviours and easy onset voice. Stage 3: Self-monitoring and carry over.

Specific considerations During therapy with children with cleft palate/VPD it is important to evaluate not only the effect of therapy on voice but also on speech in general. Changes in voice may result in perceptual changes in resonance, i.e., following voice improvement a child with cleft/VPD may sound more hypernasal. Therefore, it is necessary to monitor all speech parameters, and proceed to instrumental evaluation of palate function if necessary. Although therapy approaches used in children with cleft palate/VPD are not different to the general paediatric population, when working with this group it is important to think about how voice therapy fits into their overall management plan. For example, the timing of therapy may need to be considered in cases where the child is awaiting surgery to address VPD. Voice therapy may need to be deferred if secondary surgery to improve velopharyngeal function is imminent. Other interventions such as grommet insertion and alveolar bone graft (ABG) may be a priority at a given time; therefore, it is important that voice therapy is seen as part of a multidisciplinary treatment programme. Within the context of speech and language therapy, input for articulation deviations (particularly non-oral articulation such as glottal and pharyngeal articulation patterns) may sometimes precede voice therapy, as they influence perceptual judgements of speech and may even contribute to the voice disorder, arising from laryngeal hyperfunction (Witzel, 1995).

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Voice and VPD management issues It is often a dilemma in clinical practice when patients with both VPD and voice disorders are concerned, which to manage first: the voice pathology or the velopharyngeal dysfunction? Given the limited research on voice therapy in children with voice problems secondary to VPD it has been suggested to correct the VPD first, prior to considering voice therapy (McWilliams et al., 1990). Similarly, other authorities have argued that voice therapy will not be effective until velopharyngeal adequacy is achieved (Cavalli, 2011; PetersonFalzone et al., 2010; Boone, 2004). Surgical removal of vocal fold nodules without improving the VP mechanism has been suggested as ineffective. Improving the VP mechanism can be associated with improvement in vocal fold pathology; however, it does not always eliminate it (McWilliams et al., 1973). Whether or not vocal pathology is present, close monitoring of voice symptoms is important and, if they are significant, intervention may be appropriate. As most voice disorders in children have a significant behavioural component, behaviour modification techniques need to be employed together with parent and family education for modification of voice use and elimination of vocal abuse (D’Antonio & Scherer, 1995). In terms of everyday clinical practice, children with cleft palate/VPD presenting with persisting dysphonia will benefit from referral to an ENT clinic for further investigations. Collaborative management is essential and should be based on vocal/laryngeal findings as well as status of velopharyngeal mechanism. Management decisions can be complex for this patient group and although it is generally advocated that the VPD is managed prior to treating the voice disorder, it is important to establish the likely causes of dysphonia particularly in cases of laryngeal pathology that may need prompt intervention.

• Research

The need for close collaboration between voice and cleft/VPD specialists became evident in a survey by Bowden (2005) who looked into management of dysphonia in 14 UK cleft centre sites. The reported incidence of dysphonia was between 5% and 10% of all cleft/VPD patients at the time of the survey. However, only 5 sites had a policy for the management of children with voice problems and VPD. Ten out of 14 sites had access to specialist speech and language therapist/ENT clinics and 3 out of 14 had a therapist specialising in voice and cleft.

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Conclusion This chapter has highlighted the importance of including evaluation of voice in the assessment of individuals with cleft palate/VPD. It has also identified the need for collaboration between cleft palate and voice specialists in management where voice and velopharyngeal disorders coexist. Finally, the lack of clear research evidence in this area suggests that inter-centre studies of voice problems in individuals with cleft palate/VPD would be useful. This data may potentially redefine the prevalence of dysphonia in this population. In addition, consensus on assessment protocols, data collection and data analysis will help to more accurately describe the voice profile of these children, measure treatment outcomes and make recommendations for best practice.

• Case study: John

John was born with a unilateral cleft lip and palate. At his routine 18-month speech and language therapy assessment at the cleft centre, it was noted that he presented with hypernasality and nasal emission accompanying sounds. Review by the ENT surgeon also revealed retracted eardrums, so grommets were inserted. He was reviewed again in SLT aged 2 years 3 months and then again at 2 years 8 months and 3 years. He continued to present with significant nasal emission and hypernasality. He had appropriate sound placement apart from palatalisation of /t d/ and occasional backing of /d/ to /g/. Given his persisting VPD, a lateral X-ray (videofluoroscopy) was performed aged 3 years. This revealed a short palate with anterior muscle position and consistent VP gap. He subsequently had a palate re-repair together with a Hynes pharyngoplasty. John’s 6-month post-operative speech assessment showed mild hyponasality and no nasal airflow errors. Dysphonia was first noted at the age of 3 years 8 months and was becoming more noticeable. John was seen in a paediatric voice clinic aged 5 years. The following factors were noted as placing him at risk for dysphonia: mouth breathing due to mild hyponasality causing throat dryness, fluctuating hearing loss, asthma (using inhaler), limited fluid intake during the day, some tension and shyness, and a history of VPD. On the GRBAS voice assessment, John’s voice presented as: Grade 1, Roughness 1, Breathiness 1, Asthenia 0, Strain 0. The voice SLT advice consisted of: auditory training to develop awareness of voice, contrasting high/low pitch, contrasting rough and smooth voice. He was encouraged to drink more water during the day and was encouraged to also breathe through his nose.

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Due to an increase in asthma symptoms his medication was changed and following this his mother reported fluctuating voice symptoms. He was referred back to the voice clinic aged 7 years. His mother reported that when regularly using his inhaler, his voice symptoms were worse but improved when he stopped using it. Hearing was normal at that time. Voice assessment revealed mild vocal strain but otherwise normal voice at the time of the voice clinic appointment. Unfortunately, John did not tolerate endoscopy. The ENT surgeon queried presence of vocal fold nodules. Further voice SLT was offered but the family decided not to take it up. This case highlights the synergistic effect of various risk factors contributing to dysphonia. It is interesting that John’s dysphonia was noticed after his VPD was resolved. It is difficult to know whether his past history of significant VPD contributed to his voice problems or whether it was mostly a product of a number of coexisting risk factors.

• Case study: William

William was referred to the cleft palate service at the age of 6 years 10 months. He presented with occasional mild hypernasality and had no history of cleft or other medical issues. However, William had a history of dysphonia from a very young age, with vocal fold nodules diagnosed aged 4 years 5 months. He was described as a quiet child, with no history of shouting or abusing his voice. The voice SLT commented that his profile of dysphonia and nodules was unusual, given the absence of any obvious risk factors. William had voice SLT input for dysphonia secondary to nodules. Voice therapy helped reduce his symptoms, but he was still dysphonic at the time of the cleft SLT assessment (his voice was described as mildly rough, breathy and weak). The cleft SLT assessment revealed evidence of mild hypernasality, which was difficult to rate accurately due to his significant dysphonia. There was no nasal emission but very occasional nasal turbulence accompanying sounds. William had SLT for articulation focusing on backing and fast rate of speech but he struggled to generalise these targets. His lateral X-ray (videofluoroscopy) showed a long thick palate that elevated well, with slight anterior muscle position. There was apparent VP closure during speech but was not always firm. Nasendoscopy followed, revealing a hairline gap across the palatal plane

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during connected speech. There was good closure on syllables and single sounds; the gap was mostly evident in spontaneous speech. Palatal muscle retro-positioning was offered by the surgeon as the only surgical option to address his mild VPD. It was explained to the family that it was difficult to establish whether the very mild VPD was the cause for William’s vocal fold nodules and so resolving it would not necessarily change his voice profile. The parents were not keen on a surgical procedure, especially involving an uncertain outcome for his voice. There was liaison between the cleft and voice SLT, who agreed that a further course of voice therapy was the most appropriate course of action at the time. His palate function continued to be monitored. This case highlights the following: William is a child with an early history of dysphonia, even though there were no associated risk factors that would explain his voice profile and presence of vocal fold nodules. However, the presence of mild VPD and slight articulation difficulties together with fast rate of speech could have been contributory factors. The dilemma in this case is how best to manage his dysphonia, given that an operation on his palate was not certain to improve his voice, his VPD was minimal and inconsistent, and his parents were not keen on a surgical option that was not certain to eliminate dysphonia. The decision for both SLTs involved (voice and cleft/VPD) was whether to continue with voice therapy and then reassess the need for palate surgery, or suggest surgery first, given that past voice therapy was unsuccessful in eliminating his voice symptoms. The voice SLT was unsure why William did not improve with voice therapy and so surgery for VPD may be the only option in the future. However, given the above diagnostic and management uncertainties, it was agreed to follow a conservative approach and continue with voice therapy while closely monitoring his palate function.

References Aronson, A. (1990). Clinical Voice Disorders: An Interdisciplinary Approach, 3rd Edn. New York: Thieme Inc. Boone, D. (2004). G. Paul Moore Lecture: Unifying the disciplines of our voice Smorgasbord. Journal of Voice, 18(3), 375–386. Bowden, M. (2005). VPI and voice problems. Presentation at the National Special Interest Group for Cleft Palate/Craniofacial Anomalies. Derby.

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Carding, P., Carlson, E., Epstein, R., Mathieson, L. & Shewell, C. (2000). Formal perceptual evaluation of voice quality in the United Kingdom. Logopedics, Phoniatrics and Vocology, 25, 133–138. Carding, P., Roulstone, S., Northstone, K. and the ALSPAC study team (2006). The prevalence of childhood dysphonia: A cross sectional study. Journal of Voice, 20(4), 623–630. Cavalli, L. & Pinkstone, M. (2005). Managing the complexities of dysphonia and VPI. Presentation at the National Special Interest Group for Cleft Palate/Craniofacial Anomalies. Derby. Cavalli, L. (2011). Voice assessment and intervention. In S. Howard & A. Lohmander (Eds), Cleft Palate Speech: Assessment and Intervention. Chichester: Wiley-Blackwell. D’Antonio, L., Muntz, H., Province, M. & Marsh, J. (1988). Laryngeal/voice findings in patients with velopharyngeal dysfunction. Laryngoscope, 98(4), 432–438. D’Antonio, L. & Scherer, N. (1995). The evaluation of speech disorders associated with clefting. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach. St Louis: Mosby. De Bodt, M., Ketelslagers, K., Peeters, T., Wuyts, F., Mertens, F., Pattyn, J. & Van de Heying, P. (2007). Evolution of vocal fold nodules from childhood to adolescence. Journal of Voice, 21(2), 151–156. Grunwell, P. & Sell, D. (2001). Speech and cleft palate/velopharyngeal anomalies. In A. Watson, D. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Guyette, T., Sanchez, A. & Smith, B. (2000). Laryngeal airway resistance in cleft palate children with complete and incomplete velopharyngeal closure. Cleft Palate-Craniofacial Journal, 37(1), 61–64. Hamming, K., Finkelstein, M. & Sidman, J. (2009). Hoarseness in children with cleft palate. Otolaryngology – Head and Neck Surgery, 140(6), 902–906. Henningsson, G., Kuehn, D., Sell, D., Sweeney, T., Trost-Cardamone, J. & Whitehill, T. (2008). Universal parameters for reporting speech outcomes in individuals with cleft palate. Cleft Palate-Craniofacial Journal, 45(1), 1–17. Hocevar-Boltezar, I., Jark, A. & Kozeli, V. (2006). Ear, nose and voice problems in children with orofacial clefts. The Journal of Laryngology and Otology, 120, 276–281. Imatomi, S. (2005). Effects of breathy voice source on ratings of hypernasality. Cleft PalateCraniofacial Journal, 42(6), 641–648. John, A., Sell, D., Sweeney, T., Harding-Bell, A. & Williams, A. (2006). The cleft-audit protocol for speech: A validated and reliable measure for auditing cleft speech. Cleft PalateCraniofacial Journal, 43(3), 272–281. Kuehn, D. & Moller, K. (2000). Speech and language issues in the cleft population: The state of the art. Cleft Palate-Craniofacial Journal, 37(4), 348–383. Ladefoged, P. (1993). A Course in Phonetics, 3rd Edn. Orlando: Harcourt Brace.

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Leder, S. & Lerman, J. (1985). Some acoustic evidence for vocal abuse in adult speakers with repaired cleft palate. Laryngoscope, 95, 837–340. .

Lee, E-K. & Son, Y-I. (2005). Muscle tension dysphonia in children: Voice characteristics and outcome of voice therapy. International Journal of Pediatric Otolaryngology, 69, 911–917. McWilliams, B., Lavorato, A. & Bluestone, C. (1973). Vocal cord abnormalities in children with velopharyngeal valving problems. Laryngoscope, 83, 1745–1753. McWilliams, B., Morris, H. & Shelton, R. (1990). Cleft Palate Speech, 2nd Edn. Ontario: BC Decker. Mathieson, L. (2001). Greene and Mathieson’s The Voice and its Disorders, 6th Edn. London: Whurr Publishers. O’Connor, J. (1973). Phonetics. London: Penguin. Peterson-Falzone, S., Trost-Cardamone, J., Karnell, M. & Hardin-Jones, M. (2006). The Clinician’s Guide to Treating Cleft Palate Speech. St Louis: Mosby. Peterson-Falzone, S., Hardin-Jones, M. & Karnell, M. (2010). Cleft Palate Speech, 4th Edn. St Louis: Mosby. Sell, D., Harding, A. & Grunwell, P. (1999). GOSSPASS’98: An assessment for speech disorders associated with cleft palate and/or velopharyngeal dysfunction (revised). International Journal of Language and Communication Disorders, 34(1), 17–33. Trani, M., Ghidini, A., Bergamini, G. & Presutti, L. (2007). Voice therapy in paediatric functional dysphonia: A prospective study. International Journal of Paediatric Otorhinolaryngology, 71, 379–384. Van Lierde, K., Claeys, S., De Bodt, M. & Van Cauwenberge, P. (2004). Vocal quality characteristics in children with cleft palate: A multiparameter approach. Journal of Voice, 18(3), 354–362. Witzel, M. (1995). Communicative impairment associated with clefting. In R. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach. St Louis: Mosby. Wood, K.S. (1971). In Peterson-Falzone, S., Hardin-Jones, M. & Karnell, M. (2010). Cleft Palate Speech, 4th Edn, (p. 221). St Louis: Mosby. Wyatt, R., Sell, D., Russell, J., Harding, A., Harland, K. & Albery, E. (1996). Cleft palate speech dissected: A review of current knowledge and analysis. British Journal of Plastic Surgery, 49, 143–149.

7 Principles of Early Intervention Sandra Treslove

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Aim of this chapter • To explore what is known about the impact of a cleft palate (CP) on babble and early speech in babies, and how this may affect later phonetic and phonological development. • To discuss why, in the absence of any firm evidence-base, targeted early intervention from the age of 18 months could benefit children with cleft palate. • To describe an early speech intervention approach. • To provide ideas and resources for multisensory input modelling therapy.

Introduction Cleft palate, with its associated risk of hearing loss, is widely acknowledged to place babies at risk of delayed and/or atypical speech and language development (Persson et al., 2012; Chapman, 2011; Morris & Ozanne, 2003; Chapman et al., 2001; Harding & Grunwell, 1996). While the debate surrounding optimum timing for palate repair is ongoing (Lohmander et al., 2012; Hardin-Jones & Jones, 2005; Rohrich et al., 2000), speech and language therapists in clinic are faced with the more immediate question of how best to support children born with a cleft palate with their developing speech, language and communication skills. Most traditional speech therapy models focus on direct intervention from about the age of three, once children are able to demonstrate a readiness to comply with articulation or phonological therapy activities. However, as our understanding of the impact of a cleft on early phonetic and phonological development grows, so do arguments in favour of intervention in the prespeech and early speech stages (Scherer et al., 2008; Harding-Bell & Bryan, 2008; Golding-Kushner, 2001; Russell & Harding, 2001). Alongside this, there is also a growing body of literature exploring psycholinguistic approaches to therapy (Stackhouse & Wells, 1997) allowing us to analyse the impact of cleft palate on a child’s underlying speech processing skills. In this context, the argument to work with a speech sound system as it develops and to intervene to destabilise atypical speech patterns before they become firmly established (Russell & Harding, 2001) is a persuasive one.

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“The goal of early intervention programmes is to diagnose and treat speech and language problems at the earliest time, in the hope of minimising the effects of early delays and disorders. For children with cleft palate, an important additional purpose of early intervention is to prevent the development of compensatory articulation errors and to treat them if they do occur” Golding-Kushner (2001), p.43.

Vocal development in babies with and without a cleft palate Pre-speech to speech: The typically developing infant The seminal works of Oller and Eilers and colleagues in the 1980s and 1990s contributed significantly to the understanding of stages of vocal development in the typically developing infant from birth through to early words. A fourstage model is described (Oller et al., 1999): • Phonation stage: Consisting of glottals and quasi-vowels. Quasivowels are so-called because they are produced with the vocal tract at rest and without articulation of the lips, tongue and jaw. • Primitive articulation stage: This typically occurs by 2–3 months of age. It is described as ‘gooing’ by the American authors, but is perhaps more familiar as ‘cooing’ in the UK. Infants begin to move the vocal tract (i.e., articulate) while vocalising. At this stage, the airstream is still nasal. • Expansion stage: Described as full vowels, raspberries, squeals, growls and marginal babbling. Marginal babbling is characterised by the transition from a consonant-like sound to a postured full vowel. • Canonical stage: Canonical babble is defined by a rapid transition from consonant-like sounds to vowel-like sounds, producing wellformed syllables such as /bi/ /dada/ /nunu/. These are considered the building blocks for spoken language, with adult-like timing in their rhythmic structure. At this stage, usually established by ten months of age, parents tend to shape their infants’ vocalisations to

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word-like purposes, for example /dada/ may have no meaning for the infant but be interpreted as meaning ‘daddy’ by an adult. By this stage, movement of the palate determines whether the airstream is oral (palate closed) or nasal (palate open). In the first two stages (Phonation and Primitive articulation), the structure of the larynx and size of the tongue is such that vocalisations are by default nasal, and early sounds are made using a high tongue position and back tongue contacts.

• Did you know …

All babies are obligate nose breathers At birth, the larynx has a different shape and structure from that of the adult. It is higher and more horizontal which means that the epiglottis and soft palate are brought close together. At the same time, the tongue is relatively large and fills the oral space. Consequently, infants are forced to breathe through their nose (Peterson-Falzone et al., 2006; Harding & Grunwell, 1996). The increased range of sounds heard during the expansion stage is enabled by anatomical changes in the oral cavity. At around 4–6 months of age, the larynx begins to lower and a space emerges between the epiglottis and the soft palate. This creates for the first time the ability to separate the oral and nasal cavities. Once the soft palate is able to rise to seal off the nose from the mouth, it is possible for the infant to produce oral consonants. These are sounds formed by the build-up and release of intraoral pressure, familiar to SLTs as the obstruent class of speech sounds (Ladefoged, 2005). As Chapman and Willadsen (2011) point out, this is a key developmental step since the nasal/oral contrast is present in a vast majority of languages. At this stage, too, typically developing babies begin to acquire greater control of tongue movements to engage in vocal exploration. Peterson-Falzone et al. (2006) describe what they term the ‘alveolar take-over’ at around the age of six months when anterior labial and alveolar consonants begin to predominate, and glottal stops decrease in frequency.

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Pre-speech to speech: The infant with a cleft palate Up until the age of about four to six months, when early vocalisations are by default nasal, the presence of a cleft palate has little impact on sounds made by infants. It is only once the larynx lowers that early sound differences begin to emerge. Without the ability to seal off the nose from the mouth, it is not possible to build sufficient intraoral pressure to produce the obstruent class of speech sounds, and the ability to mark nasal and oral contrasts is lost. The ‘alveolar take-over’ described at around the age of six months for babies without a cleft palate is not observed in children with a cleft palate and glottal stops persist (Peterson-Falzone et al., 2006).

• Note

Key features of early babble in children born with a cleft palate: Restricted range of sounds – mostly nasals, glides and approximates Dominance of sounds made with the back of the tongue Fewer sounds made with the front of the tongue and lips Predominance of glottal stops Fricatives, if present, are likely to be glottal or pharyngeal. One study found that over half of babies with an unrepaired cleft palate had not reached the canonical babbling stage by nine months of age (Chapman et al., 2001). This is significant because a delay in the onset of canonical babble has been proposed as a marker of later speech and language disorder (Oller & Eilers, 1999; Oller et al., 1998). For children with cleft palate, it is important to consider not only the impact of an altered anatomy during the crucial stages of exploratory sound play, but also the potential impact of fluctuating hearing loss on the quality of sounds they are able to hear (see Chapter 5).

• Note

A psycholinguistic perspective Psycholinguistics is an approach which explores the underlying cognitive processes which may give rise to a child’s speech and literacy difficulties (Stackhouse & Wells, 1997) and is explored more fully in Chapter 9.

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Throughout the present chapter, specific reference will be made to psycholinguistic hypotheses in relation to the emerging speech sound system of infants and toddlers with a cleft palate. Relating an unrepaired cleft of the palate to the speech processing model (Stackhouse & Wells, 1997), the presence of the cleft can be interpreted as representing a primary motor execution deficit. The range of motor programs that can be carried out correctly is restricted by the infant’s atypical anatomy. In addition, the presence of fluctuating hearing loss would impact on peripheral auditory processing on the input side of the model.

Early words: The typically developing toddler In children without a cleft palate acquiring language typically, first words emerge at around the age of one. For a period, they will continue to produce non-meaningful babble alongside these first words (Chapman & Willadsen, 2011). Widely accepted research in the field of lexical selectivity shows that toddlers will select words to say that start with sounds already existing in their consonant inventory. They are less likely to acquire words in their early vocabulary containing sounds they have not yet acquired accurately (Willadsen, 2012; Stoel-Gammon, 2011; Estrem & Broen, 1989; Schwartz et al., 1987) (see Table 7.1).

Early words: The toddler with a cleft palate In the UK, most children will have had surgery to repair the palate (known as primary palate repair) by the end of their first year of life. Following surgery, their vocalisations tend to progress towards more typical patterns (Russell & Albery, 2005). Glottal and pharyngeal articulations decrease, and there is an increase in velar plosives (Russell & Grunwell, 1993). However, their first words continue to be influenced by their pre-repair speech sound repertoire; unlike their peers without a cleft, who are likely to target more words with an initial plosive, fricative or affricate, they are more likely to target words beginning with vowels, nasals or approximants (Willadsen, 2012; Estrem & Broen, 1989).

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Table 7.1 Sounds favoured word initially in toddlers with and without cleft palate Toddlers without a cleft palate

Toddlers with a cleft palate

Plosives Fricatives Affricates

Vowels Nasals Approximants

• Did you know …

Babies with a cleft palate who are attempting to make plosive consonants close to or soon after palate repair have improved speech and language outcomes (Chapman, 2004; Chapman et al., 2003). Following primary palate repair, some children acquire typical speech without the need for SLT intervention. However, a study of 212 children with cleft palate found that as many as 68% did require speech and language therapy (Hardin-Jones & Jones, 2005). For the SLT working with the child at this stage, two key considerations emerge: • Following primary palate repair, it is possible that the palate mechanism is still compromised and intraoral pressure continues to be reduced. This is known as velopharyngeal dysfunction (VPD) (see Chapter 2). There may also be a hole (fistula) remaining between the oral and nasal cavity, resulting in reduced intraoral pressure and potentially in audible nasal escape through the hole (Harding & Grunwell, 1996). In this instance, further surgery to repair the palate is likely to be necessary. Speech therapy for cleft speech characteristics can begin prior to further palatal surgery, and also has a diagnostic role in the evaluation of the VPD itself (Sell & Grunwell, 2001).

• Terminology

The term Cleft Speech Characteristics (CSCs) is used to describe a pattern of errors frequently heard in the speech of children with a cleft. Very often, articulations are made further back in the vocal tract, where a greater sense of pressure may be obtained (Trost, 1981). See Chapter 3.2 for a summary of CSCs.

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• Speech sound errors can also persist in children where primary palate repair has been judged to be successful. This means that cleft speech cannot be interpreted as an articulatory disorder alone. Rather, atypical speech sounds from babble (acquired at a stage when the palate is unrepaired) become absorbed into the early phonology of the child. This is the origin of the description of cleft speech as an articulatory disorder with phonological consequences (Grundy & Harding, 1995).

• Psycholinguistic note

Looking again at the speech processing model, (Stackhouse and Wells, 1997), primary palate repair can be interpreted as having resolved the motor execution deficit. However, in children where speech sound difficulties persist, it appears that new motor programs continue to adopt the atypical patterns established prior to repair. In turn, these atypical motor programs will impact on phonological representations when the child starts to acquire these with the onset of meaningful speech at around 12 months of age (Calladine, 2009).

Advice and early management: The role of the speech and language therapist Birth to 18 months In the UK, babies born with a cleft lip and palate are referred to one of 10 multidisciplinary centres. On the whole, early speech and language development advice and routine monitoring are provided by specialist SLTs from these centres. However, service models vary and this aspect of care may be delivered by a community-based SLT with additional knowledge of cleft palate. The speech and language therapist will aim to ensure that families have a broad understanding of typical speech, language and communication development. He or she is likely to recommend a range of activities to encourage the development of language and communication skills, such as singing nursery rhymes, patterning back speech-like sounds made by the baby, encouraging imitation of face, tongue and lip movements, and playing auditory awareness games (Russell & Albery, 2005). Other advice will be more specific to babies with a cleft lip and/or palate. This may include:

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• Information about the role of the palate in speech • Information about the change in sounds parents may hear following surgery to repair the palate • A description of what the SLT listens for in the speech of the child as he or she develops over time. This broadly covers how well the palate is working and how the child is making different speech sounds • Advice to encourage parents to enjoy sound play activities with their child • An explanation of why families should avoid reinforcing glottal or pharyngeal sounds (Russell & Albery, 2005).

• Note It is hypothesised that use of glottal and pharyngeal sounds (i.e., non-oral sounds) arises from a subconscious attempt by the child to compensate for loss of pressure at the velum by using articulators lower down in the vocal tract (Trost, 1981). Parents are advised not to imitate any ‘throaty’ or effortful sounds made by their child, but to respond by modelling more gentle sounds. Families may be encouraged to make animal and transport noises without excess effort in the throat, for instance a lion to make a soft ‘rah’ rather than a throaty ‘grrr’, a pig to say ‘oink’ rather than snorting. • Gentle blowing activities using very light objects such as cotton wool or feathers, or gently blowing on the back of the baby’s hand. This is to raise awareness of oral rather than nasal airflow (Russell & Albery, 2005). • Information about the role of hearing in speech and language development, and the increased incidence of glue ear in the cleft palate population (Peterson-Falzone et al., 2006). SLTs may discuss with parents about the importance of attending audiology review appointments even when they feel that their child’s hearing is good, as there may be no obvious symptoms such as discharge or discomfort (see Chapter 5).

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18 months to 3 years Most regional cleft centres offer a routine speech, language and communication assessment between 18 months and two-and-a-half years of age. It is as a result of this assessment or follow-up review that concerns may be identified and a referral to the child’s local SLT service made. It has been shown that SLTs are able to accurately identify those children with cleft palate who will go on to require speech sound therapy from as young as 17 months of age (Hardin-Jones & Chapman, 2008). Given these specific speech sound difficulties identified in young children with a cleft palate, it has been suggested that this in itself, rather than more generalised language delay, should be the target of early intervention: “Because early phonological and lexical development appear closely related to one another, therapy for young toddlers with cleft palate may be more productive when expansion of their consonant inventory is seen as a primary goal of early intervention and not as a by-product of language intervention” Hardin-Jones & Chapman (2008, p.90). However, many providers of early years speech and language therapy offer group models of service delivery for children in this age group; for instance, language programmes such as parent–child interaction or Hanen™. It is acknowledged that such intervention may be well suited to a child with a cleft palate where additional language or social communication needs have been identified. At the present time, there is little evidence in the literature to support any specific model of early intervention (RCSLT Clinical Guidelines, 2005). Indeed, there is no evidence to support specific models of intervention for children with a cleft palate of any age (Bessell et al., 2013). At this stage, then, in the development of speech and language therapy intervention, it is valid to turn to psycholinguistic processing and a hypothesis testing approach. What follows is an exploration of those psycholinguistic principles and how they have been interpreted to inform a non-directive, input modelling therapy approach.

A psycholinguistic approach to early intervention for speech In the context of early speech intervention for children with a cleft palate there are two guiding psycholinguistic principles:

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Figure 7.1 The four anchor points of the speech processing model Adapted from Stackhouse, J. & Wells, B. (1997). Children’s Speech and Literacy Difficulties: A Psycholinguistic Framework. London: Whurr Publishers.

1. Working on input can strengthen output processing. This is because the different levels of input and output processing work together as a system rather than in isolation, so that activating relatively stronger levels of processing can help strengthen weaker ones (Rees, 2001). Input processing to strengthen output processing The concept of providing repeated models of speech sounds with no expectation of output from the child forms the basis of several therapy approaches, such as auditory bombardment (Hodson & Paden, 1991) and auditory input therapy (Lancaster, 2009). These describe the use of repeated models of phonological targets and contrasts presented in play activities with children. The approaches allow time for a child to reflect on qualities of sounds with the hypothesis that this will strengthen phonological representations and subsequently improve the accuracy of output skills. In addition, since working on input makes no demands on the speech output of the child, it adapts readily to use with very young children.

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2. Using a ‘bottom-up’ approach. There are two dimensions to the speech processing model: ‘top-down’ processing which utilises previously stored linguistic information, and ‘bottom-up’ processing which does not (Stackhouse, 1997). ‘Bottom-up’ approach It can be helpful to remember the four key anchor points of the speech processing model, as described by Stackhouse & Wells (1997) – see Figure 7.1. Let’s start from the premise that atypical speech sounds, here cleft speech characteristics, result from incorrectly stored linguistic information. In order to prevent the motor programs that trigger these from being stimulated when a sound is heard, the child must perceive that what they are hearing is something new. If a target sound is presented in such a way that no existing linguistic information is activated, it allows for new, more accurate motor programs to be created. In other words, sounds that are innovative, or ‘novel’ will trigger bottom-up processing and by-pass incorrectly stored linguistic information. This principle has been described more fully by Harding-Bell and Bryan (2008) as a key component of their multisensory input modelling therapy.

• Note

What happens when a child hears an innovative speech sound? Theoretically, an innovative sound will be recognised as a speech sound at speech/non-speech discrimination in the model. As it is novel, it will not trigger matches within phonological recognition or motor programming, and so will be stored temporarily in phonetic discrimination. If the sound is then heard repeatedly, a match will be made in phonetic discrimination. In turn this should stimulate motor programming, with the goal of creating a new, accurate motor program.

Multisensory input modelling therapy (MSIMT) The principle of using novel/adapted speech sounds to stimulate bottom-up processing forms a central tenet of multisensory input modelling therapy

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(MSIMT) described by Harding-Bell and Bryan (2008). The term multisensory describes the inclusion of not only auditory but also visual and kinaesthetic cues. This allows the child to reflect fully on different qualities of a sound, such as the manner or place of articulation. The key principles of MSIMT are: • Use of adapted or novel speech sounds • Frequent repetition of speech sounds • Multisensory presentation • Engaging with parents to enhance frequency and accuracy of input models at home – including videotherapy. At the present time there is no published literature describing or evaluating the use of MSIMT or different models for its delivery. A demonstration and teaching DVD is available (Harding-Bell & Bryan, 2008) and there has been one small, unpublished study (Calladine, 2009). Nonetheless, the underpinning psycholinguistic principles are widely accepted (RCSLT, 2005) and the approach continues to be the subject of clinical interest and evaluation.

Delivering early speech sound intervention: Working within a community service As previously discussed, referral to community SLT services typically occurs following centre-based routine assessment at about 18 months to two-and-ahalf years of age and sometimes earlier. Assessment will have identified areas of concern including limited speech sounds and/or cleft speech characteristics such as backing or use of glottals. What is described here is intended as a guide for SLTs working with toddlers with cleft palate in community settings. The resources and therapy ideas are presented not as a finished product but rather to raise awareness of an input-only approach and to stimulate creativity in its delivery to children and families.

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Beginning therapy Establishing aims and outcomes Aims for early speech sound intervention are: • To encourage sounds made with the lips and front of the tongue • To encourage gentle sounds requiring oral air pressure • To prevent cleft speech characteristics from becoming established in the child’s phonological system. Early speech sound intervention can be seen as, at least in part, preventative. Measuring outcomes is therefore challenging as results are not fully apparent until the child has acquired a more established phonological system. It is therefore proposed that it is acceptable to measure the outcome in relation to the ability of a parent to deliver consistent models. For a period of intervention, one aim and outcome might look something like this: Aim: For child to receive consistent, multisensory models of a range of targeted speech sounds in a play situation. Outcome: Child’s parent has demonstrated the ability to model targeted sounds consistently to their child at the final therapy session, using a multisensory, non-directive approach in play.

Additionally, parental self-rating scales and questionnaires can be valuable in providing a qualitative measure of change pre- and post-intervention (RCSLT, 2006).

Which target sounds should I select first? As a general rule, voiceless targets are selected initially as these can be modelled with no constriction at the glottis and with least requirement for oral pressure. For this group of children, it may therefore be appropriate to ignore developmental norms and focus instead on voiceless and fricative sounds (see Table 7.2) as these are likely to be easier for the child to produce at a later stage (Russell & Albery, 2005).

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Table 7.2 Target selection Assessment

What to target

Suggestions

Absence of alveolar sounds

Sounds using tongue tip and blade

Dentalised alveolar plosive [t̪] Interdental fricative [θ]

Glottal/pharyngeal substitutions

Voiceless sounds with minimal oral pressure

Anterior fricatives e.g., [ɸ] [f] [ʃ] [θ] Aspirated bilabial plosive [pʰ] Dentalised alveolar plosive [t̪]

Active nasal fricatives (ANF)

Voiceless oral fricatives

E.g., If child’s target /s/ is realised as an active nasal fricative (ANF) try modelling interdental fricative [θ] If target /f/ is ANF, model bilabial fricative [ɸ]

How many targets should I select? This will be informed by the listening and attention skills of the individual child, and how responsive families are to the principles of the therapy. Clinical experience suggests that between two and four target sounds work effectively for most children under the age of three.

How do I make targets multisensory? 1. Auditory cues • Ensure that sounds are presented repeatedly. How frequently a sound must be repeated to trigger a positive match in the temporary ‘phonetic discrimination’ store is unknown, but it follows that the more the sound is heard, the more likely a match will be made. Harding-Bell and Bryan (2008) recommend 20 repetitions of each sound in each activity. Realistically, this number is likely to vary from session to session and activity to activity, depending on the level of interest from the child.

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• Use soft contacts and minimum oral pressure. Present sounds gently, as a child may imitate any hint of strain or force. Close proximity to the child is recommended, to maximise the quality of sound input the child will receive. • Vary modelled volume and intonation. Whispering is recommended as a way to engage a child’s listening and attention (Harding-Bell & Bryan, 2008). 2. Visual cues • Draw the child’s attention to how a sound is made. Consider this when adapting sounds to make them novel for the child (see Table 7.2). For example, adapting a sound from an alveolar to dental or interdental position increases visual feedback about how the sound is made. Draw the child’s attention to what you specifically want them to notice, for example “Look – can you see my lips? They are very round – /ʃ/.” • Give the adapted sound a label/visual symbol, such as a ‘bubble wobble’ for [pʰ] or ‘seahorse sound’ for /ʃ/. A range of suggested images are provided on the Resource CD. • Gestures can also provide a visual cue to accompany a symbol or picture. For instance, if a family pet eats canned food, associate the sound of the opening can with [t̪] as a novel sound, accompanied by a ring-pulling gesture. You could call this the ‘dog food’ sound. Cued articulation can also be readily incorporated into input modelling (Passy, 1983). 3. Tactile cues • The airstream accompanying oral sounds provides a tactile cue. Let the child feel your breath on the back of their hand as you model the sound. On occasion, try holding your nose, too, to show that all the air comes through your mouth and not down your nose. • Make therapy activities more tactile by using gestures or sensory play, such as modelling clay, marbles, and feathers.

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Figure 7.2 Hand stroking input activity

• Top Tip

Harding-Bell and Bryan (2008) demonstrate a hand stroking technique whereby the therapist traces their finger along the back of the child’s hand while modelling the sound. This can be extended to putting the sound before or after a vowel as described in Chapter 8 (see example on Resource CD). Not all children will tolerate the sensation of being touched, but for those who do, it is a simple, calming activity which can be easily replicated at home (Figure 7.2).

How do I engage families in therapy? Since frequency, consistency and quality are key elements of an input approach, it is vital to work closely with families to ensure carry-over of the therapy at home. The concept of working on input with no demand for output may contradict all parental expectations of what should constitute speech therapy as the temptation to encourage output is a strong one. Families who understand the rationale for an input-only approach are more likely to engage in activities between SLT sessions. It is also important to explain to parents that there is no expectation that

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speech sound errors will have resolved by the end of therapy episode. Parental expectations will need to be carefully managed, or they may be left with feelings of disappointment that the therapy has not worked.

• Note

A note on language and parent/child interaction: The focus of this intervention is speech sound development. Nonetheless, opportunities may arise for the SLT to offer wider language and communication advice to the family, depending on what they observe. MSIMT itself can be interpreted as promoting language and parent–child interaction, for instance parents are encouraged to participate regularly in shared play activities, to select games and objects which are of interest to the child, and to use a nondirective approach.

What are the advantages of making a DVD recording? Making a DVD recording of the therapy session (‘Videotherapy’) is recommended by Harding-Bell and Bryan (2008). It can be a useful tool to: • Provide the child with the opportunity to watch modelled sounds repeatedly and accurately in between SLT sessions • Remind families how to make the sounds • Remind families of the therapy approach such as frequent repetitions of the same sound and not asking the child to repeat the sounds • Enable other family members/carers to watch the SLT and child together and so involve the wider family in therapy • Amplify the sound, which is useful where there may be inconsistent hearing loss • Potentially stimulate silent rehearsal (Harding-Bell & Bryan, 2008). See Table 7.9 (Practical considerations for using DVD) at the end of this chapter. At the current time, there is no published evidence to support the use of a visual recording in early speech intervention. Some parallels can perhaps be

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drawn with the use of video modelling to facilitate play and social interaction skills for people with autistic spectrum disorders, for which there is a weak but growing body of evidence (Axe & Evans, 2012; Mason et al., 2012; Cardon & Wilcox, 2011).

What sequence does the therapy follow? MSIMT follows a familiar sequence for SLTs working on acquisition of speech sounds: 1. Sounds in isolation. 2. Sounds in sequences (e.g., CV, VC, CVC). 3. Sounds in familiar real words.

Is it necessary to hear a change in output to move up through the sequences? This is an input-only approach; therefore, a change in output is desirable in the long term but not an expectation in the short term. The decision to move on will be made when the child has received repeated, consistent models of targeted sounds/words over a period of time. Calladine (2009) described each step lasting for two therapy sessions. However, this is in no way prescriptive. If a child has not received consistent, good quality input at step one, consider staying at the single sound level of modelling for longer. Conversely, where a child spontaneously begins to use a targeted sound accurately, you might consider modelling that sound with a vowel and then in real words.

Step One: Single sound input Table 7.3 suggests ways to present single sounds to a child and their family through play. As you model the target sounds, consider the following principles:

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• Use soft, gently stretched sounds. • Be very clear to show how you make the different sounds, by describing the shape of your lips or the movement of your tongue. For example: “[ʃ] Can you see how round my lips are when I make the seahorse sound? Look, they’re the same shape as the seahorse”. “[t̪] my tongue is very gently tapping. [t̪] [t̪]. Can you hear the sound? Look how my tongue is moving – nice and softly”.

• Research

Calladine (2009) suggests that this shaping procedure is an important component of the modelling approach, with the potential to optimise correct output practise at a later stage. She cites studies by Scherer et al. (2008) and Chapman et al. (2001), suggesting that correctness of output practice, rather than frequency, has an important role in early speech development.

• Hold the pictures or toys close to your mouth so that the child can see and hear how you make the sound at the same time. • Let your child feel the air from your mouth as you gently make the sounds – sitting close to them will help this. • At times, try gently holding your nose as you make the sound. Explain to the child that the air is coming through your mouth. Refer to it as a ‘mouth’ sound.

Use a dental tongue position to provide greater visual feedback than alveolar placement

/t/

Continues over the page

If the child is using a backed target for the alveolar plosive, then using a dental position and introducing the sound as ‘novel’ offers the potential to create new motor programming

How to model as a novel sound

Target phoneme

Useful resources/tips • Hand gesture to mimic the dripping water • Drawn outline of tap and drips – child colours in drips as you model the sound. Alternatively, the child dabs sponge-paint/stamper for each drip • Draw lots of water drops on a page – the child points to each spot as you model the sound • Song: Adapt ‘I hear thunder’ to: ‘I hear raindrops I hear raindrops – [t̪ t̪]. Hark don’t you? Hark don’t you? Listen for the raindrops – [t̪ t̪] see my tongue make raindrops – [t̪ t̪]. I’m wet through – so are you’. Keep the rhythm steady and slow and use the accompanying hand gesture each time you model the sound • This is the sound of the ladybird’s legs gently tapping as it walks along the table/up the arm of the child • Draw pictures of ladybirds – colour in/paint spots together as you say the sound • Ladybird resources such as shape cutters, finger puppets, and wind-up toys are readily available from toy shops and catalogues. Search for preschool ladybird resources on the internet • There are plenty of pre-school books featuring ladybirds, such as Ten Little Ladybirds, a counting book by Melani Gerth and Laura HuliskaBeith (Gullane’s Children’s Books, 2008). Touch the 3D ladybirds as you say the sound

Novel sound label and visual cue Dripping tap

Ladybird

Table 7.3 Input modelling therapy ideas: Single sounds

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Model very softly and gently as [pʰ]. Golding-Kushner (2001, p.78) recommends gentle overaspiration to extend the open air-flow with no involvement at the glottis

Whisper the /ʃ/ sound and use very rounded lips to give a soft, novel quality to the sound

/p/

/ʃ/

Table 7.3 Continued from preceding page

• Show the child how you stretch your lips to look like the seahorse. He makes a soft and gentle sound as he moves through the water • Cut out paper templates of seahorses and decorate them with glitter (say /ʃ/ as you shake the glitter) and/or feathers (see if you can make the feather gently move as you say the sound) • Leave them with the family to make a seahorse mobile using a hanger and cotton thread before your next visit •Cut out a tissue paper seahorse and watch it move as you make the sound

• Use a hand gesture for the bouncing space hopper, saying [pʰ pʰ] as it gently bounces • Table space hoppers can be purchased in novelty gift shops. Alternatively, use other objects such as a small balloon or pom-pom instead • Try using the space hopper or pom-pom as a marker in games such as ‘Snakes and Ladders’. Say the sound as you move the number of spaces. Remember to keep providing feedback about how you are making the sound as you play the game • As a carry-over suggestion, demonstrate a game where you gently move the space hopper or pom-pom up a staircase one step at a time. Recommend that families do this whenever they climb the stairs to bed – keep the sound soft and gentle – don’t want to wake anyone!

Space hopper

Seahorse

• Catch a bubble on the wand (Tip: Don’t economise on bubbles – the cheap ones rarely work well enough to catch on the wand) • Use Cued articulation (Passy, 1983) for /p/ as a gesture to draw the child’s attention to how you are making the sound with both of your lips

Bubble wobble

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Model as an interdental sound [θ] to increase visual feedback and reduce the oral pressure needed to make the sound successfully

Use either labio-dental or bilabial model Keep your model soft and stretched

/s/

/f/

• Present the sound as being how a baby snake learns to hiss. The baby snake has to stick his tongue out and gently blow. Show that holding your nose makes no difference to the sound. Let the child feel your airflow on the back of their hand • Try modelling snakes out of play dough or pipe cleaners. Gently run your fingers along the snakes as you say the sound • Leave a shoe box with the family as a snake house and ask the family to decorate it before your next visit • Snake socks can be fun to make and are very tactile • The internet has a wealth of resources (try searching ‘Snake craft for kids’). A snake is, however, easy to draw – a piece of paper and some coloured crayons provide plenty of opportunity for sound play without any preparation at all • Let the child feel the air coming out from a balloon as you gently release it while modelling the sound. Try holding it close to their ear so they can hear the sound. Many children also enjoy the sensation of the air tickling the soles of their feet • This sound is the dragon gently puffing as she slowly flies along. Use your arms as wings, or a toy dragon if you have one. Try modelling a very elongated sound as she comes in to land • Gently stroke pictures of dragons in books as you say the sound. For example, That’s Not My Dragon a touchy-feely book by Fiona Watt and Rachel Wells (2011) London: Usborne • Download a picture of a dragon and laminate it. Punch holes in it to make a dragon lacing card. Say the sound as you/the child threads a shoe lace in and out of the holes

Baby snake

Leaky balloon

Dragon

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Step Two: Sounds in sequences Begin with multiple repetitions of the single sound before introducing it within a speech sound sequence.

• Note

Why recap the single sound immediately before introducing the sound in a sequence? By this stage, it is hypothesised that frequent exposure to a new sound will have created an accurate motor program for it. By exciting this motor program immediately before introducing the new sound in unfamiliar words, the aim is to maximise the likelihood that a match with the new sound will be made. With each multisensory presentation of the new word, motor programming signals are strengthened, increasing the likelihood that the new correct sound motor program will be used to create an accurate motor program for the whole new word (Calladine, 2009).

Table 7.4 Sounds in sequences Child’s targets

Theme

Example Nonwords (Character names)

Activities

[t̪] [pʰ] [θ]

Ladybird family

Daddy ladybird [єt̪i] Mummy ladybird [uθ] Baby ladybird [ipʰ]

Lots of greetings as you introduce the characters ‘Hi Eti!’ Stick spots on the ladybirds as you repeat its name Colour in different parts of the ladybird as you say its name Create a ladybird world with shoeboxes and tissue paper, etc

[f] [θ]

Dragon and Fifi the dragon another /fifi/ and her character friend [pʰipʰə]

Colouring/sticking/drawing activities Introduce a giant (‘fee fi fo fum’) (check that the child is familiar with the phrase from the fairy tale first) Pop-Up dragon game (characters take turns to play) That’s Not My Dragon by Fiona Watt & Rachel Wells (Usborne, London). Use the refrain ‘That’s not Fifi …’, etc.

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Aim for simple combinations such as CV/VC or CVCV to create either unfamiliar real words or non-words. Many of the ideas outlined in Table 7.3 lend themselves to the creation of non-words in therapy activities, for example by creating character names (Table 7.4). Cartoon or animation characters that the child is unfamiliar with can be useful here, too (e.g., Popeye, Tellytubbies). Tips for creating non-words/unfamiliar real words: • Names which repeat the sound can be useful, e.g., Toto, Sissy, etc • Aim for low pressure sounds in the other word positions. /l/ /h/ and /w/ are useful • Be guided by sounds the child can already make. For instance, if you are aiming for [t̪] and the child can say /p/ then use [pɔt̪] (or ‘Pete/ Peter’ if the family say this is ‘new’ to their child) • If you feel a child has significant VPD, aim to place the target sound word finally. Should the child attempt output, there is a greater chance of success in the motor execution stage if the sound is at the end of the word (Russell & Albery, 2005).

Recommendations Remember to maintain the feedback about different qualities of the sound. For example, “Here comes the dragon [fː fː] (wings flapping). She’s called /fifi/. Did you hear the dragon sound in her name? /fifi/ I can feel the air on my hand – can you?” At this stage, you can incorporate consonant-vowel (CV) and vowelconsonant (VC) activities too. For instance, the child fishes for a sound picture from Step One, then a vowel picture. /i/ /u/ and /a/ work well as they are visually contrastive (use any vowel picture symbols you have available, or there are samples provided on the Resource CD). The child puts them either before or after the sound. Model the combination that the child has made. Keep your models very gentle and stretched. The hand-stroking technique (Harding-Bell & Bryan, 2008) described previously is useful at this stage. If the child finds the sensation calming, recommend hand-stroking as an activity parents can do each night just before bedtime.

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Table 7.5 Sounds in real words Target 1 [t̪]

Activity idea

Single sound repetition [t̪]

Sheet of ladybird pictures: Ask child to touch each picture as you model the sound

Sound in word [єt̪i]

Daddy ladybird arrives. Remember his name? [єt̪i] Repeat as you gently bounce him around the table

Introduce target words Pet, cat, rat, parrot, wait

Goes to buy a pet – will he buy a cat or a rat or a parrot? Feely bag activity

Target 2 [pʰ]

Activity idea

Single sound repetition

Give out swords for pop-up pirate. Say [pʰ] as you give each one out. “There’s the bubble wobble sound”

Sound in word [ipʰ]

Baby ladybird [ipʰ] arrives and wants to join in game

Introduce target words Up, push, pop, poppy

Pop-up pirate game – use for push, pop and up Any stacking game – up up up Bubbles – pop pop pop – wait – pop pop pop Making paper poppies

Step Three: Sounds in real words To move on to this stage, the SLT should feel confident that the child is receiving good quality, consistent input of targeted sounds and words. Families will be able to demonstrate that they are engaging the child in sound play activities and, where applicable, regularly watching the DVD together. Begin by recapping single sounds, then the targeted unfamiliar/non-words before introducing familiar words (Table 7.5).

Recommendations SLTs will no doubt have plenty of ideas for activities at the stage, such as Kim’s game and feely bags. Alternatively, use familiar stories such as The Three Little Pigs with words like ‘huff’, ‘puff’ and ‘wolf’.

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Provide word pictures for families so that they can play simple games such as posting, hiding cards around the room, or imitating actions such as ‘wipe’, ‘hop’, ‘sew’, and ‘pat’. Popular animations such as Peppa Pig™, Postman Pat™ and Toy Story™ can be very stimulating for this age of child, and there are plenty of downloadable resources freely available too.

How many real words should I introduce? Again, there is no firm answer, but it will vary depending on assessment of each child, their language skills and their attention levels. In the Calladine (2009) study the number of target words ranged from 5–13. One sound was introduced at a time and moved through each step before introducing a second. Harding and Bryan (2001) advocate modelling a range of sounds and words with each child at a session. The decision making for precise numbers of targets and manner of inputting will vary from SLT to SLT and from child to child.

Resources to leave with families between visits To ensure that the child receives input of the targeted sounds between therapy sessions, it is particularly important to engage families in the therapy activities. The following are recommendations that may assist this:

Figure 7.3 Multisensory book (Reproduced with permission of Katie Jaines, SLT, who suggested the idea)

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• Leave plenty of picture symbols to enable carry-over of activities such as pairs games, feely bags, posting and fishing, between SLT visits • Provide written advice to remind the family about the key principles of the therapy (in particular, regular, accurate modelling of target sounds) and suggested play activities. A sample is included on the Resource CD • If it has been possible to make a DVD of the session, leave this with family. Agree with the family how frequently they will aim to watch it • Ensure that the family knows they need to engage actively in sound play activities, and do not simply rely on the DVD. Explain that the DVD provides good visual and sound cues, but the child also benefits from feeling how the sound is made • Provide a home practice diary to record times in between sessions when the family has either watched the DVD or played games. Make sure that you ask to see this at the next session • Try leaving a book or game with the family on loan and demonstrate how it can be used for sound play activities. Counting books can be useful; encourage the child to touch the pictures as the adult models the sounds. A good example is Ten Friendly Fish by Debbie Tarbett (Little Tiger Press, 2008).

• Top Tip

Make a multisensory book to leave with the family containing an object for each sound, the picture symbol and a close-up photograph of the therapist or parent saying the sound. This is a useful way of engaging families, as well as boosting multisensory cues.

• Note

Key ‘take home’ messages for parents: • Present target sounds softly and gently • Bring objects/symbols up to your mouth when modelling sounds • This is input only; output is not expected at this stage • Use ‘Watch’ ‘Listen’ and ‘Feel’ rather than ‘Say’.

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Frequently asked questions

What do I/the family do if a child attempts a sound incorrectly?

Do not request correction (Harding-Bell & Bryan, 2008). Aim instead to draw the child’s attention to the specific features of the target that contrast it to their attempt (Calladine, 2009). For instance, “When I make the sound, I use both of my lips – like this – watch.” If the child continues to copy but is not achieving the target sound, encourage them to let their tongue/lips have a rest and to watch/listen to you instead.

What if they copy the sound accurately? Let the child know that they have made the sound well, and give them specific feedback on phonetic features, for instance “Your tongue gently tapped your teeth when you made that sound – I saw it move.”

• Research

Gardner (2006) demonstrated that the quality of feedback given to children receiving therapy for speech difficulties had a direct impact on outcomes, and that training of others resulted in positive change. On this evidence, ensure that parents understand to provide specific feedback to their child (“Good – you’re watching my lips really closely”) rather than imprecise praise (“Well done – that was good”).

What is the model for delivering the therapy? As previously discussed there is no prescribed model for delivering early speech sound intervention, including MSIMT. In order to meet the requirements of service provision followed by many community services, the following guide may be helpful: • 6 × 45-minute speech therapy sessions within 3 months (i.e., approximately once a fortnight) • Where possible, video the therapy session to optimise carry-over at home

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• Therapy sessions to take place in the home or clinic setting • Therapy to be delivered by SLTs or SLTAs with appropriate support from the cleft centre SLT.

• Note Why 6 sessions? Achieving the correct ‘dosage’ for speech therapy input is always controversial. Even discounting availability of resources, decision-making around frequency of input is more likely to rely on clinical experience and service models than evidence of efficacy or effectiveness (Law et al., 2003.) The outline here is designed to fit within the model of ‘blocks’ or episodes of therapy which continues to be adopted by many paediatric speech and language therapy services in the UK.

Which setting is most appropriate – home or clinic? Offering home visits allows SLTs to build rapport with the family, gain insight into their routines and daily life, and observe the communication between parent and child in the child’s natural setting. This enables recommendations for carry-over of activities to be more readily individualised, such as being able to demonstrate activities with the child’s own toys and books. Given competing demands on therapist time, however, packages combining home and clinic visits can work effectively.

Can I use this approach with older children? The approach outlined is intended for children with a cleft aged approximately 18 months to 3 years. However the core principles can be applied to older children, for instance those who are reluctant to talk in therapy, where output may be compromised by significant VPD, or where progress has plateaued. MSIMT is also recommended as suitable for children with non-cleft speech difficulties (Harding-Bell & Bryan, 2008).

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• Case study: Mary

This case study presents an example of early speech sound intervention for a 2-year-old with a cleft palate. She was reluctant to talk and there was considerable anxiety in the home about her speech.

Background Mary transferred to the cleft centre at age 20 months. She was born with a wide cleft palate associated with Pierre Robin sequence, which was repaired at age 12 months. She had a history of mild hearing loss associated with glue ear for which a ‘watchful waiting’ approach had been taken. She was slow to acquire early words, reportedly having only a few single words at 18 months. She was seen at home for SLT review at the age of 2 years 1 month. At this point, her mum described that her vocabulary was really beginning to expand. Table 7.6 Sample of Mary’s speech Word

Target

Realisation

House

/haʊs/

[aʊ] and [aʊn̥͋]

Bus

/bʌs/

[b̃ʌs̪] and [b̃ʌn̥͋]

Sun

/sʌn/

[ɱʌɱ͊ ]

Doggy

/ˈdɒɡi/

[ˈɡɒɡi]

/tu/

[ɡu]

/splæʃ/

[p̪æŋ̥͋]

Car

/kɑ/

[ɡɑ]

Bat

/bæt/

[b̃æx]

Sky

/skaɪ/

[ɡaɪ]

Peppa

/ˈpɛpə/

[ˈp̪ɛp̪ə]

Two Splash (imitation)

m

ɱ

ɱ

Target

SIWI

SFWF

-



p

-



b

-

-

f

-

-

v

ɱ͊

n

n

Table 7.7 Mary’s consonant realisations - Age 2:1

-

-

l

s̪,x

ɡ

t

-

ɡ

d

n̥͋, s̪

ɱ

s

-

n̥͋

z

ŋ̥͋

-

ʃ

-

-

ʧ

-

-

ʤ

-

-

y

-

ɡ

k

-

-

ɡ

-

ø

h

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At the first visit, Mary was shy and unwilling to talk but a small speech sample was obtained using a Peppa Pig™ felt board. See Tables 7.6 and 7.7.

What were the key features of Mary’s speech? • Resonance: Mixed hyper- and hyponasality. Mary’s Mum felt that hyponasality was not typical and related to Mary having a cold. This did not entirely mask hypernasality as /b/ was realised as [b̃]. • Nasal emission: Inconsistent nasal emission was noted, but nasal congestion meant that this could not be accurately judged. • Labiodental articulation of target /p/ and /m/ possibly related to Mary’s small lower jaw although she demonstrated potential for bilabial closure (‘bat’ and ‘bus’ in the sample). • Mary’s speech sound system was unstable, with variable productions of target sounds. • /s/ was occasionally produced as an oral target but more often as a turbulent active nasal fricative. She was happy to let me gently hold her nose while she made a noise like a snake, and doing so stopped the sound abruptly. • Early phonological processes were affecting both place and manner of articulation, e.g., backing (predominantly to velar) and frication (i.e., replacing target plosives with fricatives, e.g., ‘kite’ as [kais] and [kaix]). What were the risk factors for Mary? • Mary’s hypernasality and nasal emission suggested persisting velopharyngeal dysfunction. Relating this to the psycholinguistic framework, this marks a primary deficit in motor execution, putting her at risk of secondary deficits in motor programming and motor planning. • As she had a history of glue ear, it is fair to assume variable hearing levels could impact on the stability of her peripheral auditory processing skills. • Cleft speech characteristics existed in her repertoire and may have become more firmly established in her phonological system over time. • Mary was shy and reluctant to imitate adult models.

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Rationale for intervention at this age • Mary’s phonological system was unstable; cleft speech characteristics, such as the active nasal fricative, were emerging but were not established into her phonology. • Mary’s mother was anxious about her speech. She was increasingly aware of the ‘snorting’ sounds in Mary’s speech that made her sound different from her peers.

The programme Three adapted target sounds were selected (Table 7.8). Table 7.8 Early targets for Mary Target

Symbol

Rationale

[θ]

Baby snake

Mary’s production of /s/ was unstable (most often realised as an active nasal fricative). A novel sound will have no match in phonological recognition so will not trigger existing, incorrect phonological representation, i.e., a bottom-up approach [θ] provides a more visual cue than /s/ [θ] is a lower pressure sound than /s/ so can be modelled with minimum effort and no constriction at the glottis

[t̪]

Drippy tap

Mary’s realisation of /t/ was also unstable (backed to palatal or velar and realised as a fricative rather than plosive). As above, adapting the sound to the dental place of articulation makes it novel and provides a more visual model of how the sound is made

[pʰ]

Bubble wobble with cued articulation

Mary’s labiodental placement of [p̪] was hypothesised to be an articulatory consequence of her small lower jaw. Bilabial closure was achievable so the emphasis was on providing repeated models of bilabial closure

Typical phrases to accompany these sounds included: “Can you see my tongue when I make the baby snake sound? The air feels tickly on the back of my hand.”

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“The water is making a soft, gentle sound. Look out for my tongue – [t̪ t̪ t̪] .” “Look at my lips when I make the bubble wobble sound – [pʰ pʰ pʰ].” Mum took a DVD recording of the SLT demonstrating sound play activities with Mary at each visit.

Activities Single sounds: These included posting or fishing and pairs games with the symbols, making plasticine snakes, colouring in the picture symbols while the SLT modelled the sounds, and wobbling bubbles on the wand. Books were loaned to the family each session to encourage sound play activities between visits. For example, Ten Tiny Tadpoles by Debbie Tarbett (Little Tiger Press, 2006) for repeated sound [t̪]. Non-words/unfamiliar real words: Small toys were provided and given made-up names targeting the sounds [θ], [pʰ] and [t̪]. Explicit reference was made linking the target sounds to the words, for example “Listen for the baby snake sound when I say /bєθ/ .” Familiar words: These were introduced slowly, beginning with /t/ and /p/. The toys had a tea party and chose their food with the carrier phrases “I eat …” and “I will not eat …” (a pie, a pea, an apple, a pear, a hat, a rat, a boot). Books at this stage included Mr Magnolia by Quentin Blake (Red Fox, 2010) which rhymes words ending in /ut/ and the ‘That’s Not My’ series (Usborne Touchy-Feely board books, London.) Simple board puzzles (such as Postman Pat™, Toy Story™) were also loaned to the family, with specific words identified to model, e.g., hat, cat, pat, boot.

Early results Mary clearly enjoyed the activities, and is reported to have asked to watch the DVD in between visits. She spontaneously moved to attempting to copy the sounds and, as her attempts were accurate, this was not discouraged. She spontaneously

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imitated a bilabial plosive for [pʰ] on the first visit, and was quickly able to follow the adult model of placing this before or after a vowel, then into real words. By session 4 she had mostly generalised bilabial [pʰ] into her everyday speech. Two new sounds were introduced on the 5th session – /f/ and /ʃ/. Most significantly, the active nasal fricative began to be replaced by the oral interdental target /θ/ in spontaneous speech, so reducing the distinctive ‘snorty’ sound that had so characterised Mary’s speech. Once Mary was readily attempting the sound in isolation, both with and without nose holding, and then beginning to use it in words, she was provided with workbook-type resources for /s/. Removing all pressure to make speech sounds gave Mary confidence and she was then willing to attempt output of the sounds. Mary’s mother was highly engaged in the therapy, and reflected that she had changed from asking Mary to say a word to modelling it accurately and repeatedly instead. Dad was away in the Services for part of the time, but even he was recruited, successfully modelling speech sounds via webcam.

Mary’s speech at age 3 years At age 3, a year after intervention began, Mary came to the palatal investigation clinic. By this time she had had a total of six sessions with an SLT and five with an SLTA. She had also participated in a block of five phonological awareness sessions with the local SLT service, including activities such as rhyme awareness and syllable clapping. Speech assessment indicated that her speech sounds were age appropriate and the active and backing processes had resolved. Turbulence and hypernasality were now the most prevalent features and videofluoroscopy indicated a consistent small gap between her palate and the back of her throat during speech. Subsequently, she underwent surgery to improve closure of the palate against the back wall of the throat (pharyngoplasty). This successfully eliminated hypernasality and turbulence, and Mary required no further speech therapy intervention. In the course of a year, Mary had acquired an accurate speech sound system, including affricates /ʧ/ and /ʤ/. This had been achieved despite the presence of velopharyngeal dysfunction and suspected glue ear.

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What would have happened had Mary not received input at this early age? • Her phonology may have stabilised accurately without any intervention at all • Early cleft speech characteristics may have become firmly established in her phonological system • Phonological or articulation therapy could have been offered when she was older. In this single case, it is suggested that early speech sound intervention successfully destabilised cleft speech characteristics and encouraged more accurate ones. It is possible that early intervention using multisensory input modelling was successful in this instance because glottal stops, which are known to be resistant to therapy, were not a significant feature of Mary’s early speech.

There are many components to the intervention described, such as high intensity models of accurate speech sounds, multisensory presentation, videotherapy, regular play activities with a parent. Firm conclusions about what aspect of the intervention made the difference, if any, cannot be drawn. For that, early speech sound intervention using input modelling techniques needs to be more systematically researched.

Conclusion This chapter has sought to discuss early speech sound intervention in children with cleft palate, presenting a rationale for its use whilst also acknowledging the limited research evidence available. It has referred to one specific approach, Multisensory Input Modelling Therapy, and outlined therapy ideas for children who have cleft speech characteristics but who are too young to begin phonological or articulation therapy.

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Table 7.9 Practical considerations for DVD recordings of therapy Recommendation

Reason

Record the activities in short sections (five minutes each)

This makes it easier for parents to select different sections to play to the child

Record yourself directly facing the camera rather than in profile (Calladine, 2009)

This means the child can see the movement of your mouth clearly when the DVD is played back to them

Ask a parent to check the framing of the DVD as you record

This will help ensure that you and the child stay within the frame of the camera

The frame should be close enough so that your mouth movements can be clearly seen

Briefly check the DVD after your recording; if your face cannot be clearly seen, try recording an additional short section ‘direct to camera’ without involving the child. Use the opportunity to repeat the sounds frequently, relating them to the picture symbol card, e.g., “Can you see how round my lips are when I make the seahorse sound? [ʃː ʃː]. Just like in this picture. I can feel the air on the back of my hand [ʃː ʃː] – it’s warm and tickly”

It is advisable to ask families to sign a written agreement they will not reproduce the data on any social networking site or other electronic media

The DVD is left with families so there is the potential that clips could be reproduced in the public domain without the consent of the SLT. Legal guidance may be sought from your employer

References Axe, J.B. & Evans, C.J. (2012). Using video modelling to teach children with PDD-NOS to respond to facial expression. Research in Autism Spectrum Disorders, 6(3), 1176–1185. Bessell, A., Sell, D., Whiting, P., Roulstone, S., Albery, L., Persson, M. & Ness, A. (2013). Speech and language therapy interventions for children with cleft palate: A systematic review. The Cleft Palate-Craniofacial Journal, 50(1), 1–17. Calladine, S. (2009). Multi-sensory input modelling therapy: Intervention for young children with cleft palate. MSc Dissertation, University of Sheffield, UK. Cardon, T.A. & Wilcox, M.J. (2011). Promoting imitation in young children with autism: A comparison of reciprocal imitation training and video modelling. Journal of Autism and Developmental Disorders 41(5), 654–666. Chapman, K.L., Hardin-Jones, M., Schulte, J. & Halter, K.A. (2001). Vocal development of 9-month-old babies with cleft palate. Journal of Speech, Language and Hearing Research, 44, 1268–1283.

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Chapman, K.L., Hardin-Jones, M. & Halter, K.A. (2003). The relationship between early speech and later speech and language performance for children with cleft lip and palate. Clinical Linguistics and Phonetics, 17(3), 173–197. Chapman, K.L. (2004). Is presurgery and early postsurgery performance related to speech and language outcomes at 3 years of age for children with cleft palate? Clinical Linguistics and Phonetics, 18(4–5), 235–­257. Chapman, K.L. (2011). The relationship between early reading skills and speech and language performance in young children with cleft lip and palate. Cleft Palate-Craniofacial Journal, 48(3), 301–311. Chapman, K.L. & Willadsen, E. (2011) .The development of speech in children with cleft palate. In S. Howard and A. Lohmander (Eds), Cleft Palate Speech. Chichester: WileyBlackwell. Estrem, T. & Broen, P.A. (1989). Early speech productions in early language acquisition. Journal of Speech, Language and Hearing Research, 32, 12–23. Gardner, H. (2006). Training others in the art of therapy for speech sound disorders: An interactional approach. Child Language Teaching and Therapy, 22(1), 27–46. Golding-Kushner, K.J. (2001). Therapy Techniques for Cleft Palate Speech and Related Disorders. New York: Thomson Delmar Learning. Grundy, K. & Harding, A. (1995). Developmental speech disorders. In K. Grundy (Ed.), Linguistics in Clinical Practice (pp.329–358). London: Whurr Publishers. Harding, A. & Grunwell, P. (1996). Characteristics of cleft palate speech. European Journal of Disorders of Communication, 31, 331–357. Harding-Bell, A. & Bryan, A. (2008). The use of multi-sensory input modelling to stimulate speech output processing. A teaching and demonstration DVD produced by Addenbrooke’s NHS Trust and East & North Hertfordshire NHS Trust. Hardin-Jones, M.A. & Jones, D.L. (2005). Speech production of pre-schoolers with cleft palate. Cleft Palate-Craniofacial Journal, 42(1), 7–13. Hardin-Jones, M. & Chapman, K. (2008). The impact of early intervention on speech and lexical development for toddlers with cleft palate: A retrospective look at outcome. Language, Speech and Hearing Services in Schools, 39, 89–96. Hodson, B. & Paden, E. (1991). Targeting Intelligible Speech: A Phonological Approach to Remediation. Austin, TX: ProEd. Ladefoged, P. (2005). A Course in Phonetics, 5th Edn. Boston: Heinle. Lancaster, G. (2009). Implementing auditory input therapy. In C. Bowen (Ed.), Children’s Speech Sound Disorders. Chichester: Wiley-Blackwell. Law, J., Garrett, Z. & Nye, C. (2003). Speech and language therapy interventions for children with primary speech and language delay or disorder. Cochrane Database of Systematic Reviews 2003, Issue 3. Art. No.: CD004110. DOI: 10.1002/14651858.CD004110.

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Lohmander, A., Friede, H. & Lilja, J. (2012). Long-term, longitudinal follow-up of individuals with unilateral cleft lip and palate after the Gothenburg primary early veloplasty and delayed hard palate closure protocol: Speech outcome. Cleft Palate-Craniofacial Journal, 49(6), 657–671. Mason, R.A., Ganz, J.B., Parker, R.I., Burke, M.D. & Camargo, S.P. (2012). Moderating factors of video-modeling with other as model: A meta-analysis of single case studies. Research in Developmental Disabilities, 33(4), 1076–1086. Morris, H. & Ozanne A. (2003). Phonetic, phonoloical and language skills of children with a cleft palate. Cleft Palate-Craniofacial Journal, 40(5), 460–470. Oller, D.K., Eilers, R.E., Neal, A.R. & Cobo-Lewis, A.B. (1998). Late onset canonical babbling: A possible early marker of abnormal development. American Journal on Mental Retardation 103(3), 249–263. Oller, D.K. & Eilers, R.E. (1999). Precursors to speech in infancy: The prediction of speech and language disorders. Journal of Communication Disorders, 32, 223–245. Passy, J. (1983). Cued Articulation. St Mabyn: Stass Publications. Persson, M., Becker, M. & Svensson, H. (2012). Academic achievement in individuals with cleft: A population-based study. Cleft Palate-Craniofacial Journal, 49(2), 153–159. Peterson-Falzone, S.J., Trost-Cardamone, J.T., Karnell, M.P. & Hardin-Jones, M.A. (2006). Treating Cleft Palate Speech. St Louis: Elsevier Mosby. Rees, R. (2001). Principles of psycholinguistic intervention. In J. Stackhouse and B. Wells (Eds), (1997). Children’s Speech and Literacy Difficulties: Identification and Intervention, Chapter 2. London: Whurr Publishers. Rohrich, R.J., Love, E.J., Byrd, H. & Johns, D.F. (2000). Optimal timing of cleft palate surgery. Plastic and Reconstructive Surgery, 106, 413–422. Royal College of Speech and Language Therapists (2005). Clinical Guidelines. Bicester: Speechmark. Royal College of Speech and Language Therapists (2006). Communicating Quality 3 Guidance on Best Practice in Service Organisation and Provision. London: RCSLT. Russell, J. & Albery, L. (2005). Practical Intervention for Cleft Palate Speech. Oxon: Speechmark Publishing. Russell, J. & Grunwell, P. (1993). Speech development in children with cleft lip and palate. In P. Grunwell (Ed.), Analysing Cleft Palate Speech. London: Whurr Publishers. Russell, V.J. & Harding, A. (2001). Speech development and early intervention. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Scherer, N.J., D’Antonio, L. & McGahey, H. (2008). Early intervention for speech impairment in children with cleft palate. The Cleft Palate-Craniofacial Journal, 45(1), 18–31.

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Schwartz, R.G., Leonard, L.G., Frome Loeb, D.M. & Swanson, L. (1987). Attempted sounds are sometimes not: An expanded view of phonological selection and avoidance. Journal of Child Language 14(3), 411–418. Sell, D. & Grunwell, P. (2001). Speech assessment and therapy. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate, Chapter 16. London: Whurr Publishers. Stackhouse, J. & Wells, B. (1997). Children’s Speech and Literacy Difficulties: A Psycholinguistic Framework. London: Whurr Publishers. Stengelhofen, J. (1990). Working with Cleft Palate. Bicester: Winslow Press. Stoel-Gammon, C. (2011). Relationships between lexical and phonological development in young children. Journal of Child Language, 38, 1–34. doi:10.1017/S0305000910000425. Trost, J.E. (1981). Articulatory additions to the classical description of the speech of persons with cleft palate. Cleft Palate Journal, 18(3), 193–203. Willadsen, E. (2013). Lexical selectivity in Danish toddlers with cleft palate. The Cleft Palate-Craniofacial Journal 50(4), 456-465.

8 Speech Sound Therapy Helen Piggott

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Aim of this chapter This chapter is designed to be a practical resource for clinicians working with children who have a cleft palate. It discusses broad aims and objectives, suggesting a progression through sound development. The aim is to provide clinicians with ideas and practical support in how to plan and deliver speech ‘output’ activities where the focus is on developing new speech motor programmes. Some of the strategies discussed may be new to the community clinician, but are based on theories of articulation and phonology and will be transferable to other client groups. The development of paralinguistic skills and generalisation are given special consideration, as both can be challenging. Finally, children will also require development of their input/phonological skills to support the establishment of new sounds in their speech. These skills have been discussed in earlier chapters, in the context of the psycholinguistic model and in relation to early intervention approaches.

Setting the scene for therapy When the clinician is focusing on ‘input’ skills, the child can adopt a more passive listening role within the therapy process. When the focus of the sessions is on ‘output’, the opportunities for failure and opting out are inevitably greater. Clinicians are often undertaking therapy with young children (three years onwards) or children who are older and have had a lot of therapy support in the past. Both groups provide specific challenges in terms of compliance and achieving positive outcomes. The following are some ideas and considerations to help ensure successful and rewarding sessions for both the therapist and child. Younger children: 1. Use fun cause-and-effect toys for repetitive sound practice. Some favourites include: Manic Martians™, Jitterbugs™, Greedy Gorilla™ and Crocodile Dentist (MB Games). 2. Personalise speech practice charts with the child’s interests e.g., Postman Pat®, Mr Tumble, or the child’s pet.

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3. Go through the home practice leaflet with parents (see Resource CD). 4. Provide a clear explanation to parents (and child where appropriate) about therapy goals. Use the palate diagram (see Resource CD) to explain changes in tongue placement and airflow. 5. Use a visual timetable to work through games in the session. 6. Use a folder to keep games and worksheets together, and encourage the child to personalise this. 7. Agree a reward with the parent and child for compliance in the session, e.g., watching part of a new DVD, going to the park after the session, etc. Older children: 1. It is worth spending time exploring why an older child may be reluctant to take an active part in the therapy process. Frank discussions with parents also help to tease out potential problems. Examples of reasons why children don’t want to participate fully in therapy include: they are always pulled out of their favourite lesson; they are not concerned about their speech as their family understands them; or they find the sessions boring. Children often do not understand how unintelligible their speech is, and this can be addressed by using video and audio recordings of their speech. 2. Keep sessions fun. Older children often enjoy playing younger games in the confines of the therapy session, but wouldn’t want their friends to know they did! On the other hand, some do not want to engage in these sorts of games because they find them childish, so it is important to cater to individual preferences. 3. Older children are often quite competitive, and this can be used to motivate. 4. Tap into the child’s interests using the topics as rewards, or use them for phrase level or generalisation work. For example: Star Wars™, Britain’s Got Talent, Top Gear, The Great British Bake Off, etc. 5. Use timers to mark out practising time. There are some good commercially-available visual/auditory timers, e.g., ‘Time Tracker® Visual Timer & Clock’, which can be pre-programmed from one minute upwards. Alternatively, the good old egg timer can be used.

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6. Small Filofax™ size folders work better with older children or young adults. If the child has a tablet device, they may enjoy entering their activities onto this and downloading suitable apps. 7. Identifying a ‘goal’ to work towards is helpful. It could be a clear telephone conversation with a relative, or a speaking part in a play, etc.

Speech sound stimulability Assessment of the child’s potential for sound production is an integral part of the initial assessment, and features throughout the therapy process. Unlike children with a phonological delay, children with cleft palate, especially those with compensatory articulations, may be unable to produce even basic oral consonants in isolation. The stimulability process involves the clinician assessing the child’s ability to produce a consonant or ‘close’ consonant (in terms of place and/ or manner of articulation), initially at a single sound level. For example, a child may be unable to achieve labiodental placement for /f/, but can make a bilabial fricative (which can later be modified with therapy to /f/). The exact sounds that the clinician is trying to elicit will depend on the child’s existing repertoire of sounds, in single words and connected speech. Where there are few or no oral consonants, stimulability testing may be carried out using nasal occlusion (see section on nose-holding, p.177). Stimulability assessment will also include CV (consonant-vowel) and VC (vowel-consonant) production with those sounds that were successfully elicited at a single consonant level. The use of nonsense or non-words is recommended at this stage, because this avoids the child associating any meaning with the sounds, and therefore reverting back to their stored motor programmes and habitual compensatory productions. The clinician can provide the child with visual, auditory and tactile cues about tongue position, direction of air stream, etc. However, this initial foray is often better executed by a ‘watch me and copy’ type approach, rather than with long explanations, which may be of more use later on in the therapy process. It can be helpful, though, to give the child specific verbal and visual feedback if their production is not accurate.

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For example: “That was a really good try.” (At /f/) “I can see that your teeth are resting on your lip.” “Could you now try to let the air come out of your mouth?” “See if the air can make the string move.” “Watch how I do it again.” (Therapist models a long, soft /f/)

• Top Tips For Successful Stimulability Games • Make games fun!

• Start with sounds that the child can say, or airstream games • Use novel toys such as feathers and crazy soap • Praise any sound attempts • Use repetitive games with a clear start and finish, to elicit sounds • Link sounds or adapted sounds to novel pictures, e.g., leaky balloon /f/, train slowing down [ts͡].

It is during these sessions that parents may hear a ‘proper’ sound for the first time. This can be a very powerful experience, enabling them to realise that their child does have the potential for more accurate speech production, and providing reassurance that improvement can be achieved. Furthermore, this can also motivate parents to engage fully in the therapy process.

Nose-holding: Why, when, and how to do it The use of nose-holding, sometimes called nasal occlusion, to facilitate sound production may be quite alien to the community clinician. When starting out it can also feel rather invasive. However, it is a strategy that can be very effective when treating children with cleft palate, and when an active nasal fricative is present. Nose-holding can be used in children with cleft palate where there is concern that they are unable to produce oral pressure consonants due to velopharyngeal dysfunction (VPD) (Harding & Grunwell, 1998). Some clinicians advocate its use during sound play exercises in very young children

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(Golding-Kushner, 2001) but in the UK, clinicians are more likely to use this strategy as part of a structured therapy programme with children aged around three years upwards. The purpose of nose-holding is to facilitate an oral airstream by artificially preventing the escape of air down the child’s nose, thus enabling the child to feel the oral airstream pass across the articulators. It can be used as a diagnostic tool where there are concerns about VP function, and within therapy to facilitate production of oral pressure consonants. When the nose is occluded, production of the sound may be stronger and/or more accurate, e.g., [p͋] to [p]. The use of nose-holding also helps in the diagnosis of an active nasal fricative (see Chapter 10.2). Clinicians should have a clear understanding of why they are using nose-holding to facilitate sound production, as its use will inevitably provoke questions from parents/carers. A clear explanation and rationale can be given to parents, using the palate diagram (see Resource CD) to talk about airflow during production of oral pressure consonants. Nose-holding in the presence of VPD usually facilitates improved sound production in terms of strength and/or

Figure 8.1 A child occluding their nose in therapy

Figure 8.2 Boardmaker™ ‘hold nose’ prompt card The Picture Communication Symbols ©1981– 2010 by Mayer-Johnson LLC. All Rights Reserved Worldwide. Used with permission.

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accuracy, and therefore parents realise its value and are generally supportive of this approach. Even young children seem to appreciate how nose-holding helps them in their sound production, especially when accompanied by visual feedback. For example, a child attempts /p/ without nose-holding, and there is minimal movement of the bubble on a wand. With their nostrils occluded, they are able to wobble or even pop the bubble on production of the /p/ sound. The child should be encouraged to hold their own nose using their two index fingers placed either side of their nose, as illustrated in Figures 8.1 and 8.2. This enables the clinician to see the child’s mouth and allows the airstream to move freely, providing the child with visual feedback on their sound production.

• Top Tips For Nose-Holding • Explain to parents why nose-holding is being used • Use a ‘hold nose’ visual prompt card to remind the child • Either the adult can hold the child’s nose or they can hold their own nose • Parents and clinicians should hold their own nose when modelling sounds • Alternatively, try a soft swimming peg during exercises.

Oral/nasal contrast – When it is useful and how to introduce it A number of children will benefit from being given feedback about airflow in sound production to help them redirect their airstream more orally. This oral/ nasal contrast is taught in a similar way to any other speech-related concept such as long/short sound, back/front sound, etc. Feedback activities, such as the use of coloured feathers, provide children with a visual prompt to help them redirect the air: • Child is prompted to ‘make the air come out of your nose’ (often linked to the Mr Men® character, Mr Nosey). Then they are encouraged to ‘Make air come out of your mouth’. This time the ‘mouth’ air is linked to Miss Chatterbox from the same Mr Men® series

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• The use of nose-holding can initially assist children in directing the air more orally • Input activities (auditory discrimination) with the adult producing sounds can be useful. The child is required to sort according to whether he hears a ‘nose’ /m n ŋ/ or a ‘mouth’ /s f ʃ p t/ sound • Self-rating scales can be used showing a picture of a mouth or nose with smiley faces below. The child has to decide whether their production was oral (correct) or nasal (incorrect). This approach works well with older children and encourages a more active role in the therapy process.

• A note on oral airstream

Previous chapters have discussed that palate function is not improved by undertaking blowing and sucking activities. However, games that aim to elicit a soft oral airstream, often using nose-holding, are valuable in allowing the child to feel air across their articulators. The child can then be taught to modify this airstream using their lips and tongue to produce sounds. Oral airstream games can be taught as an effective precursor to sound development. The Oral Airstream Activity Sheet is included in the Resource CD. Ideas are intended to be used in conjunction with advice and modelling from a clinician.

Resources Sound pictures This publication does not include a specific set of sound pictures as there is a large choice available, and each clinician has their own favourite scheme. However, two particular resources are worth mentioning. The Nuffield Centre Dyspraxia Programme NDP3®, provides pictures to link to individual consonants, many CV and VC word combinations, including approximant sounds which are useful with children who have a restricted consonant inventory or predominantly a non-oral articulation pattern. This programme also provides a structured progression up to phrase level. Another picture resource is Boardmaker™, which offers huge flexibility

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within its own vocabulary, but also allows the user to import vocabulary pictures via the Internet, enabling the clinician to target vocabulary starting or ending with a specific sound that is meaningful to the child. Well-presented, colourful picture material is motivating for the child and their family to use. Laminating these resources allows them to be used time and time again, and is really worth the initial cost outlay. Younger children also enjoy using objects to represent the consonant sounds, for example, a plastic snake for /s/, and also for carryover games, a process by which the new consonant sound is established into the child’s everyday talking. There are also a number of commercially available games and books that can be used with specific ‘sounds’ in mind, for example: Usborne’s ‘That’s Not My …’ book range.

Toys for eliciting sounds and oral airstream A box of toys (The Magic Box) just for eliciting sounds and playing oral airstream games is recommended. There are lots of novel and interesting toys that can be easily sourced, for example (see Figures 8.3, 8.4 and 8.5): Feather pens: Often in the shape of birds but they come in all shapes and sizes to cater for a range of age groups. Some pens light up when you press the end. The adult, by taking control of the light, can tell the child that the air coming out of their mouth lit up the light! Blowing bubbles: Small pots are best, as they are small enough to carry around in your bag, yet reliable and easy to hold close to the child’s mouth. Kids Stuff ® Crazy Soap: Bath time fun soap. This can be squirted to form a string (like crazy string, but lighter). The string requires hardly any airflow to move and blobs can be made to jump off the end of your finger with sound production such as /p/. The best part is that when rubbed into your hand the soap disappears as if by magic, especially when the child blows on their hand!

SLT apps There are now many apps available, targeting specific sounds, for download onto a smart phone or tablet device. These can be used effectively during therapy sessions or for home practice.

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Figure 8.3 Child using Crazy Soap ‘string’

Figure 8.4 Child using Crazy Soap ‘blob’

Figure 8.5 The Magic Box – examples of feathers

Diagnostic therapy Diagnostic therapy can be used with any age group, but is most often used with younger children who are presenting with a restricted phoneme inventory and/ or compensatory articulation errors, and where VPD is suspected. Clinicians can use a period of diagnostic therapy, typically 4–6 sessions, to gather more

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information about the child’s speech, and to help them arrive at a differential diagnosis regarding VP function.

The main aims of diagnostic therapy • To explore potential for producing oral pressure consonants • To note any strategies such as nose-holding that seems to facilitate sound production • To judge the impact of other factors, such as hearing loss • To provide an opportunity to assess ‘input’/discrimination skills and how they may be impacting on speech ‘output’.

Sessions are likely to include • Explanation of the palate diagram with regards to articulatory placement and nasal and oral airflow • Oral airstream activities • Stimulability exercises with and without nose-holding • Progression onto CV and VC words where applicable.

• Note

Parents of children without a cleft palate will need clear explanations about diagnostic therapy, as they will not have routinely received information about palate function that children born with a cleft receive. There may also be anxiety associated with the possibility that their child has a ‘structural’ problem. Where VPD is suspected, it is likely that an assessment in the Palatal Investigation Clinic will be offered (see Chapter 4). If this is the case, then diagnostic therapy may also include preparation for palatal investigation. This can include role playing what will happen at the clinic. For example: sitting still on the chair; copying the words/ sentences that the clinician says; and using the microphone. Some children also have the opportunity of visiting the radiology suite prior to the clinic. Videofluoroscopy (see Chapter 4) is only really helpful in diagnosing VPD if the child can produce at least one oral pressure consonant in isolation, but preferably be able to say the sound within a CV or VC combination. Where

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children do not have any oral pressure consonants, part of the diagnostic therapy process is to ‘work up’ a selection of words containing oral pressure consonants that can be used during this procedure. Consonants chosen will depend on what is stimulable. Generally, first choice would be /p/ in isolation or the word ‘up’ and /f/ or ‘off ’.

Case studies The following case studies demonstrate the importance of diagnostic therapy, and show how the differing individual responses to this period of therapy helped the clinician arrive at a differential diagnosis and plan future management. All but one child presented in a community clinic setting.

• Case study: Ben

Ben, aged 2.1 years, presented with only a velar nasal sound /ŋ/, few vowels, and was not stimulable for other sounds due partly to his young age. He made his needs known through gestures accompanied by /ŋ/. Eye contact was age appropriate. No early feeding issues were reported. However, he was having three bottles of milk a day and had access to his dummy throughout the day and night. Ben had been referred for a hearing test. He had not cooperated for oral examination.

Diagnostic therapy Ben was seen for four sessions over a period of approximately three months. Discussions with Mum included: • Using the palate diagram, with regards to how the velar nasal sound is produced and oral/nasal contrasts • What sounds we would expect Ben to be using at his age • How prolonged use of a dummy and bottle can affect sound production as well as dentition • The importance of having a varied diet in terms of different textures for chewing, etc. Once Mum realised the potential impact that the dummy and bottle were having on speech production she worked in partnership with her Health Visitor to phase them out.

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Sessions also included • Symbolic noises especially bilabials (moo, baa, miaow) • Modelling of early language strategies to Mum such as offering verbal choices. The oral airstream activity sheet (Resource CD) was discussed and given to Mum for her to try some easy games with Ben during bath time. Ben was encouraged to play ‘open your mouth’ games, using a crocodile puppet to prepare him for oral examination.

Outcome Without the constant dummy use Ben had more opportunity to experiment with sounds. He gradually increased his repertoire of consonant and vowel sounds, especially bilabials, and began communicating using single words. As Ben was now using some oral pressure consonants and had complied with an oral examination, which did not show any obvious structural anomaly, he was discharged back to the community SLT team for follow up.

• Case study: Millie

Millie, aged 2.8 years, was born with a unilateral cleft lip and palate. She had grommets inserted at aged 2.5 years due to frequent episodes of glue ear leading to a moderate conductive hearing loss. Millie presented with nasal /m/, /ŋ/, glottal /ʔ/ and pharyngeal /ħ/ articulation. A referral for palatal investigation had been discussed with and agreed with parents. Her receptive language was age appropriate. She was linking words together but speech was only intelligible to familiar adults.

Diagnostic therapy Millie was seen fortnightly at home over a period of approximately three months. The following was included: • Sound placement for glottal/pharyngeal ‘throat’ sounds and ‘front’ bilabial and alveolar sounds was discussed with parents using the palate diagram (see Resource CD) • Nose-holding was introduced and encouraged during therapy sessions and home practice • Oral airstream games were modelled using materials from the Magic Box

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• The /p/ and /f/ sounds were introduced using novel names and objects and quickly worked up into functional words in a word final position: ‘up’, ‘hop’, ‘help’, ‘off ’, ‘leaf ’, etc., then into word initial position • Alveolar placement /t/ was introduced using a glowing toy called Mr Tongue Tap and a jelly snake for /s/. These sounds were then developed in a similar way to /p/ and /f/. During therapy sessions, toys at home were used for sound repetition. Millie’s younger 18-month-old brother was included in the games as well. Millie tolerated the nose-holding well, which was gradually phased out as she became able to control production of the new ‘oral’ sounds.

Favourite motivating toy Fairy wand – used to point to the pictures before turning them over and saying the sound or word. Outcome Millie continued with monthly support for the next three months, with parents continuing to model words and gently encourage her to use her new sounds. Good hearing levels were maintained. She went on to develop a fully contrastive sound system and did not require referral for palatal investigation.

• Case study: Sam

Sam, aged 3.2 years, presented with only /m/, /n/, and /ŋ/ sounds in his speech and prior to therapy had not been stimulable for other sounds. There was no history of early feeding problems. Oral examination showed no structural anomalies, but minimal palate elevation on /ɑː/ phonation. Sam’s mum had attended the Hanen™ programme and he had received a block of therapy targeting auditory discrimination skills.

Diagnostic therapy Sam was initially seen for three consecutive weekly sessions with his Community SLT. The following was included:

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• The function of the palate in speech production was discussed with Mum using the palate diagram (see Resource CD). Production of nasal versus oral consonants was explained within the context of Sam’s presenting speech pattern • Nose-holding was introduced during the first session. Sam was encouraged to produce an oral airstream to make the bubble wobble and the crazy soap string move • An aspirated /pʰ/ was introduced. Sam was encouraged to ‘stop the air

with his lips’ and quickly produced a /p/ sound

• Picture cards were introduced for the /m/, /p/ and /b/ sounds and sound picture links established through listening games • The concept of oral versus nasal (Mr Mouth, Mr Nose) was introduced to support correct sound production • Alongside this Sam had success in copying /f/ in isolation, introducing him to a change in both manner and place of articulation • Single sound ladders and roads as well as blending games for CV and VC (see Resource CD) were used to establish the new motor patterns. A similar approach was used with the alveolar sounds /t/ and /d/.

Favourite motivating toy Crazy Soap™ ‘blobs’ jumping off his finger when he said /p/ or /b/. Outcome Sam’s case followed a developmental pattern with the introduction of bilabial sounds first. Nose-holding was quickly phased out when it became apparent that Sam’s palate was functioning well to produce oral consonants. Listening games and ‘role reversal’ games with the puppet were introduced to ensure that these new phonological representations were laid down. Sam remains under the care of the Community SLT Team.

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• Case study: Rachel

Rachel, aged 3.3 years, presented with predominantly glottal and pharyngeal articulation and a few nasal /m/ and /n/ sounds in her speech. Mum reported early feeding problems, which improved when solids were introduced. Rachel had a history of ear infections and associated hearing loss. Grommets had been inserted at age 2.7 years. Rachel was examined by the consultant plastic surgeon and no obvious submucous cleft palate (SMCP) was found. However, Rachel’s presenting speech pattern suggested that it might be due to issues with palate structure or function. At the time that she was seen for diagnostic therapy she had already been referred to the Palatal Investigation Clinic.

Diagnostic therapy Rachel was seen once a week, for approximately six weeks, with follow up activities carried out at home. The following was included: • The function of the palate in speech production was discussed with Mum using the palate diagram (see Resource CD) • Nose-holding was introduced during the first session. Rachel was encouraged to produce an oral airstream to make the bubble wobble and the crazy soap string move. Mum was given the Oral Airstream sheet (see Resource CD) and encouraged to try some of the activities during bath time. During the second session Rachel was able to produce some oral consonants with nose-holding. • The leaky balloon picture was introduced for /f/ and the candle picture for /p/. These sounds were practised repeatedly using motivating games such as Manic Martians™. Sound picture links were reinforced through listening games • Once Rachel’s articulation at a single sound level was accurate, VC sound blending games were introduced with /f/ and /p/ • Games used included ‘fishing for a vowel’, the vowel hand game (see Resource CD), ‘roll a vowel dice’, etc.

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• Activities then moved onto CVC words ending with /p/. Pictures were used in games, e.g., hop, hope, heap, hoop, hula hoop, etc. By using an initial /h/ the glottis is open and this helps prevent glottal reinforcement • CVC words were also introduced for final /f/, e.g., huff, hoof, half, leaf, laugh, loaf. The Three Little Pigs story was used to help establish these words (off, roof, huff, puff, laugh, etc.) • Once Rachel was able to say some single sounds and simple words ending with /p/ and /f/, she practised for videofluoroscopy in the therapy room using her picture cards as prompts, and a toy microphone (see Resource CD for preparation sheet).

Favourite motivating toy Giant magnet, used to ‘catch’ pictures in talking and listening games. Outcome Results of palatal investigation showed that Rachel’s palate was of good length but had minimal movement. She underwent surgery, and the consultant noted that there was ‘low muscle volume’ in her palate. Rachel continued with therapy to address her compensatory articulation errors. She was finally discharged aged 5.2 years with clear speech and a fully contrastive sound system.

Starting therapy The first choice for modifying cleft speech characteristics (CSCs) is the traditional sequenced articulation therapy approach (Peterson-Falzone et al., 2006). This focuses on phonetic placement and shaping techniques. The aim of therapy activities, therefore, is to help the child produce and then establish new sounds, which are currently not in their phonetic inventory. To support this process the clinician can use the following cueing strategies: auditory, visual, phonetic, verbal, manual and/or tactile (Golding-Kushner, 2001). Children will find some strategies more helpful than others. Furthermore, children

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with cleft palate frequently have intermittent hearing loss and therefore the clinician cannot just rely on providing an auditory cue. Some examples of cueing strategies are listed below: • “Watch me and listen. I am doing a soft/long/quiet sound.” • Give specific feedback about what the child did well and what they need to do next to improve the sound. • Explain tongue positions to an older child using the palate diagram (see Resource CD). Talk about sound targets on a coloured ‘zone’ (e.g., red for alveolar). • Use the palate diagram to explain tongue positions to parents. Always get the parents to make the target sound themselves, and to understand what they are doing with their articulators to produce the sound. • If the child does not copy correctly, you can encourage the parent to quietly copy the child’s attempt at the sound, and to think about what they would need to change to make production the same as the target sound. The challenge for both the clinician and the parent is to convey those changes to the child in a way that they are able to understand and implement. A five stage approach has been proposed to provide an effective articulation therapy programme (Golding-Kushner, 2001; Morley, 1970). Russell and Albery (2005) summarise these steps as below: Step 1 Eliciting a target consonant in isolation Step 2 Eliciting VC and CV combinations Step 3 Eliciting single words Step 4 Eliciting short phrases Step 5 Generalisation of the new sounds into connected speech.

Step 1: Single sound production There are a number of ways of facilitating single sound production. The ‘Up Your Sleeve’ Table (see Resource CD) provides the clinician with ideas for each sound, and includes some techniques described by Russell and Albery (1994),

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Golding-Kushner (2001), Peterson-Falzone et al. (2006) and Stenglehofen (1990). Once accurate production of a sound has been achieved, it needs to be repeated many times. Hoch et al. (1986) advocate eliciting a minimum of 100 correct productions per session. This can be achieved using ‘drills with motivating toys as rewards’ (Golding-Kushner, 2001). Examples of ladder or road games for drills are given in the Resource CD. g Further Reading: The reader is directed to Golding-Kushner, K.J. (2001). Techniques for the elimination of abnormal compensatory errors. Chapter 6 in Therapy Techniques for Cleft Palate Speech and Related Disorders.

A note on the use of ‘deferred responsibility’ Deferred responsibility can be a useful tool to use with younger children who are aware that their attempts at sound production are not accurate, preventing them from trying again and being open to modifying the produced sounds. The strategy is to ‘blame’ a body part rather than acknowledging that the child said it incorrectly. Puppets can also be used in this way. Some examples are given below: When no sound is produced, e.g., child postured for /p/ only: SLT: “I think that sound might be in your tummy. Let’s try again so that Teddy can hear the sound.” When the child says an active nasal fricative instead of an oral /s/ or [ts͡] sound: SLT: “I think that sound disappeared into your nose. Your nose likes making those sounds. Let’s try again. This time, let your mouth have a turn at making the sound.” When child says a pharyngeal or glottal sound instead of an alveolar one: SLT: “I think I heard a throat sound then. Perhaps your tongue is getting tired. Could you wake up your tongue?” In each case the clinician would model the sound again for the child, emphasising the key aspect that they want the child to change.

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Step 2: VC and CV combinations Frequent accurate repetition of the target sound should be carried out until 90–100% accuracy is achieved (Golding-Kushner, 2001). Only then should the clinician proceed to blending the target sound with a vowel. Consideration should be given to which vowels to initially use. This will depend on the target consonant. For example, a forward, high /iː/ vowel would be selected to facilitate alveolar placement for /t/, rather than using a back open vowel such as /ɑː/. Nonsense or non-words are selected, to establish the new motor patterns before introducing real words. Russell and Albery (2005) describe the method of hand stroking to teach the child smooth transitions for linking vowel to consonant and consonant to vowel. A pictorial example based on this approach is shown in the Resource CD.

• Top Tip Suggested Games for Step 2: VC and CV combinations The easiest way of introducing this stage is to have a selection of the target consonant pictures, and pictures representing the vowel cards, and to try some of the following: • Roll a large dice with vowel pictures on the side • Fish for a vowel • Use Nuffield NDP3® ladder or road vowel sheets • Springy spider ‘catches’ a vowel card • Use a magic wand to point to vowel picture • Pull a vowel card out of a bag. Older children love the magnetic balls, which can be made to move together as the child blends the two sounds.

Step 3: Eliciting single words Once the child is confidently blending at a non-word level, real words can be introduced. Finding meaningful vocabulary starting or ending with the target sounds can be difficult. However, children can be effectively taught new vocabulary at home. For example, ‘heap’ is a good word for children to practise

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if they have few oral pressure consonants and final /p/ is the focus of therapy. The parent can teach the word in a meaningful way at home, by talking about the ‘heap’ of leaves in the garden, a ‘heap’ of rubbish, or the ‘heap’ of dirty washing, etc. This can then be changed to a ‘pile’ of dirty washing when the child moves on to initial /p/. Clinicians can support the child’s production of target consonants in words by continuing to provide visual cues in the form of picture sound cards, e.g., a rabbit for /f/. Preceding and following consonants are selected to support accurate production of the target sounds and, where possible, words selected should aim to develop clear words. For example, with a child who has a very restricted consonant inventory /w, h, m, n/ and where /f/ is the target sound, the clinician would introduce words containing these sounds, e.g., ‘farm, film, foam, fireman, fan, fin, phone, half, hoof, Miffy (character by Dick Bruna), wiff, woof, Wilf (Oxford Reading Tree© character), knife’ etc. As before, the aim should be 100% accuracy before moving on to phrase level.

Step 4: Phrase level The use of carrier phrases (Golding-Kushner, 2001) is an easy way to extend the length of utterance whist ensuring that the target word is produced correctly. As with single words, the aim is to build up a repertoire of intelligible speech, using only the child’s accurate sounds. Using the previous word level example, suitable carrier phrases could be ‘we are… ’, ‘are you… ?’, ‘my… ’, ‘more… ’, ‘here are… ’, ‘half a ... worm/welly/wheel/ham,’ etc. The choice of phrases is extended if the clinician decides to include words that contain sounds that are not accurate, e.g., ‘I can… ’. This does not present any problems providing parents are clear that such sounds will need to be ignored and not corrected. As with the stimulabilty games, hearing their child produce a clear phrase for the first time can give parents a huge boost. Example word lists to generate phrases are included in the Resource CD.

Generalisation When undertaking therapy with a child with a cleft palate: “The ultimate goal is to transfer or generalize, correct production to conversational speech with automaticity” Golding-Kushner (2001), p.93.

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Generalisation is probably the second most difficult milestone to achieve after the child has mastered production of the sound in isolation. Generalisation of a sound, or group of sounds, can take place whilst therapy is ongoing or may occur immediately following a block of therapy. In community settings, time spent on establishing sounds at phrase level is unlikely to be a priority when the amount of direct therapy time available may be limited. Children with delayed phonology will generally establish new sounds given time. Unfortunately, children with cleft palate, especially those with non-oral or compensatory articulation errors, will continue to use their ‘old’ speech patterns unless a structured support system is put in place to facilitate this change.

Key considerations for successful generalisation • Parents need to be clear about which sounds are being encouraged and which are not. A typical example of a misunderstanding, is where a parent is reporting that “Johnny can’t say spoon” when the therapy was aiming to target only initial and final /s/ in simple CVC words. • Help parents and other adults (or older siblings) to tune into the child’s errors themselves, otherwise they will not perceive the speech to be incorrect, and then not prompt and support change. • A step-by-step approach is useful. For example, identifying 10 key words that the child uses and encouraging those first. • Support parents to identify and agree suitable motivators for the child (and reminders for themselves). These can range from simple sticker charts right up to a special treat. • Model strategies that demonstrate how parents can support their child’s speech and the wording to use when their child says something incorrectly, including using visual prompts where appropriate. • Explain to parents why it is important that their child is given an opportunity to work out what went wrong, which sound they need to change, and how this will help carryover to be effective. The child should be encouraged to take an ‘active’ role in the process and not just copy back what the adult has modelled, e.g., “That wasn’t right, it’s not a goor it’s a door.”

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• The older the child is, the more ingrained the habit pattern will be. This will present a challenge for the clinician, as the child will require a larger support network and greater motivation in order to effect a change in their speech.

Practical ideas to support generalisation of sounds Provide a visual prompt for the child. Examples include: • A laminated card with a single sound on it (e.g., tap) that parents show the child to remind him to say the /t/ sound when talking • A laminated card which has some words on it and a picture, which can be included in a reading folder (see Resource CD) • A reward board, which is a reminder, or a reward board that has spaces for stickers or stars to be added when the adult hears the sound (see Resource CD) • A word board which has spaces for words to be fixed on with Velcro™ or Blu Tac™. Usually the child selects the words by lucky dip into a bag. Those would be the words to remember and practise that day (see Resource CD) • Involve other family members in promoting the reward board. For example, “Show Nanny how many stickers are on your snake board.” Reward correct use of sounds and ignore errors. This can be very effective with younger children. Create situations when you know the child will be successful and remember the sound, for example, sharing a book about a character they know and can say well. Then verbally praise the child when they use the sound, adding a sticker onto their board, etc. If the sound is used incorrectly outside of this time, just model the word back to the child, stressing the incorrect sound. Work on the paralinguistic skill of rate. Encourage the child to slow down their speech at set times during the day (see example later in this chapter). Agree specific times when the child has to remember their ‘new’ sounds. Gradually increase the time slot or the frequency of the slots. Time Tracker™ toys are excellent motivators for this approach. (Search the Internet for

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demonstrations of Time Tracker™ visual timer and clock and the Mini Time Tracker™.) Agree a vocabulary list with parents, e.g., 10 frequently used words, and then gradually increase this. These words could go on the visual board. Once mastered then add on another 10. Develop self-monitoring skills. The following are examples of how the adult could respond when the child says a word incorrectly: Example: Child has pervasive backing pattern. Target word in sentence “door”; child says “goor”. “Do you open a goor or a door?” “You open a goor, what’s that?” The adult could just look puzzled and the child may then repeat the word back correctly. “Pardon?” “I don’t know what a goor is?” The adult can also facilitate improved production by providing the child with a verbal prompt: “I didn’t hear a tongue tap sound that time.” “Could you tell me that again, remembering your lovely tongue tap sound?” “I think you might have forgotten the tongue tap sound that time!”

Deferred responsibility As previously discussed, this is a useful strategy to use throughout therapy especially with younger children; however, it can also be helpful in the initial stages of generalisation when the child is often forgetting to use the ‘new’ sound/s. By ‘blaming’ the child’s tongue or throat, etc., it prevents the child thinking that they have got it wrong yet again. An example might be: “I think that your tongue is tired. I didn’t hear your long snake sound. Could you wake your tongue up and get him to say that word again?”

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Establish sounds through reading activities Once the child is reading, generalisation through literacy skills can be used very effectively, often by highlighting letters that the child needs to remember to say using their ‘new’ sound.

Supporting paralinguistic skills It is also possible to bring about a change in speech intelligibility and support the generalisation of sounds by taking a more holistic approach to the child’s speech and communication. Developing the child’s paralinguistic skills of volume and rate can impact positively on speech intelligibility. Some children may have established their speech sounds quite well at a single word or short phrase level, yet are slow to generalise these sounds into everyday situations. Others use their ‘best’ talking within the confines of the therapy situation, yet school and home report ongoing issues around intelligibility. g Further Reading: Clinicians are directed to Talkabout, A Social Communication Skills Package by Alex Kelly Ltd, and Talkabout for Children 2, published by Speechmark.

t Useful Link: Talkabout publications available from Alex Kelly Ltd: www.alexkelly.biz

The chapter on ‘Talkabout the way we talk’ provides ideas and activities to support development of paralinguistic skills. An example of the stages is given below. Activities have been adapted from Kelly’s Programme. Additional visual support materials made using Boardmaker™ symbols can be devised for use with younger children (non-readers) and those with learning difficulties. Additional materials are available to buy from the Alex Kelly website. Aim: To increase the volume of the child’s speech.

1. Teach concept: loud/quiet. 2. Gross discrimination using SLT or video, i.e., ‘very loud voice, OK, very quiet voice’. 3. Activity selection, e.g., what volume would you use in different situations, e.g., assembly, cinema, church, etc?

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4. Peer review of volume, including ‘phone a friend’ or ‘phone a teacher’ cards as prompts. 5. Generalisation. Use strategies to remind the child to maintain appropriate volume, such as a visual prompt card. This approach also lends itself to any child where vocal hygiene advice is pertinent, i.e., avoiding excessive volume. It can also be used successfully with children with cleft palate, in a mainstream setting, through small group working with peers and covers a range of skills, e.g., talking too much, interrupting others, not talking enough, or using too loud a volume. Of all the paralinguistic skills, the rate of talking is probably the aspect that impacts on intelligibility most. Modifying rate of speech can be effectively used to support the generalisation of speech sounds by slowing the child’s speech down, allowing them time to think about the speech sounds that they are using, and encouraging them to engage their new speech patterns rather than default back to their habitual ones. g Further Reading: Clinicians are directed to the chapter discussing ‘Tortoise Talking’ in Practical Intervention for Early Childhood Stammering – The Michael Palin Centre Approach, by Elaine Kelman and Alison Nicholas, Speechmark (2008). This concept was originally developed by Meyers and Woodford (1992).

An example is given below. Activities have been adapted from the Tortoise Talking Programme, for use with younger children at home and within school environments. Visual support materials are employed at each level to develop understanding of the concepts, as well as providing children with a concrete prompt which can then be used in other situations. Aim: To slow down the child’s rate of speech 1. Teach concept. Read the hare and rabbit story. Sorting activities – Fast/slow transport pictures, etc. 2. Monitor adult talking (fast/OK/slow), then each other. 3. Object prompts such as bricks or shells lined up. 4. Repetitive phase activities. The girl is ____. Adult pointing to brick, then other child pointing to brick. 5. Open discussion, e.g., Top Gear (again using prompt board).

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The following visual prompts can be used in different environments. An example is given in the Resource CD (Tortoise Talking). The Wildmutt™ character from Ben Ten™ (who is slow) is used to prompt the child to slow down his speech. A row of Wildmutt™ pictures is mounted onto card and laminated. During group discussions, news time, etc., the child uses his ‘slow talking’ by pointing to the pictures (one for each word). At home, parents verbally prompt the child to “Use your Wildmutt™ talking please”, and he knows to slow down and modify his rate. In another example, a child progresses from a row of shells as prompts, to a row of gold stars stuck onto a ruler. This is placed beside her on her desk so that she can quite discreetly point to or touch the stars to slow down as she is talking. A row of mini pictures can used in a similar way, mounted on a card strip, to prompt older children to use slow talking in general conversation. As with any strategy, success often depends on the attitude and approach taken by the key adults. Ensuring that the adults understand what the goal is and why it has been targeted to support speech intelligibility is the first step to effective support.

Completing the picture on home practice The maxim ‘practice makes perfect’ is never truer than when related to speech practice. Authors vary in their recommendations to parents/carers/ teaching staff in terms of carryover of therapy activities, but it is recognised that children with a cleft palate show most progress when focused, intensive and frequent therapy sessions are provided. (Watson et al., 2001). Follow-up home- or school-based activities are essential if the child is to make progress with their speech.

Tips for successful home practice • Explain to the parent and child (where appropriate) the structure of the palate and how therapy targets fit within this. Use the palate diagram (Resource CD) to explain airflow and the different articulatory positions. • Encourage parents to take an active role within the therapy sessions. This is ‘necessary in order for the child to accept the parent in the therapist role’ (Golding-Kushner, 2001).

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• Agree frequency of follow-up sessions at home and use home practice star charts to remind and motivate. An example is included in the Resource CD. • Discuss factors that contribute to successful sessions at home. For example, working face-to-face, being in a quiet environment, etc. Some suggestions for discussion are included in the Home Practice leaflet on the Resource CD. • Encourage parents to keep all activities together and easily accessible, preferably in a folder so that they can ‘grab and go’ when it is time to practise. • Discuss and agree involvement of siblings. If the child is older, take their lead as to who participates in their home practice sessions. • Encourage parents to be honest regarding the amount of practice that has been carried out. For example: If a child has practised every day and not made progress that is very different to practising once a week and not moving forward. Similarly, the clinician should show some understanding of the challenges faced by parents trying to fit in practice around a busy home life. • Be clear about parents’ expectations from therapy. They should be realistic, but sufficiently optimistic to motivate their child.

In summary The goal for therapy intervention with children who have a cleft palate is to effect a positive change in speech, resulting in improved intelligibility and fewer breakdowns in day-to-day communication. Cleft specific techniques to facilitate sound production, such as noseholding, have been explained, as well as their application in therapy sessions. The rationale behind diagnostic therapy has been discussed and illustrated through several case studies. This chapter has described specific activities for eliciting sounds and suggested a progression from stimulabilty of single sounds through to developing words and phrases. It has also provided activities and ideas for establishing new sounds in everyday speech. The importance of the role of parents/carers in the therapy process has been emphasised, with practical examples of how to fully engage their support.

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This chapter also includes recommendations for selecting therapy materials, especially the use of novel games contained within the Magic Box.

References Golding-Kushner, Karen J. (2001). Therapy Techniques for Cleft Palate Speech and Related Disorders. NY: Delmar. Harding, A. & Grunwell, P. (1998). Active versus passive cleft-type speech characteristics. International Journal of Language and Communication Disorders, 33, 329–352. Hoch, L., Golding-Kushner, K., Sadewitz, V.L. & Shprintzen, R.J. (1986). Speech therapy. Seminars in Speech and Language, 7, 311–323. Morley, M.E. (1970). Cleft Palate and Speech, 7th Edn. Baltimore: Williams and Wilkins. Peterson-Falzone, S.J., Trost-Cardamone, J.E., Karnell, M.P. & Hardin-Jones, M.A. (2006). The Clinicians Guide to Treating Cleft Palate Speech. St. Louis: Mosby. Russell, J. & Albery, L. (2005). Practical Intervention for Cleft Palate Speech. Oxon: Speechmark Publishing Ltd. Stenglehofen, J. (1990). Working with Cleft Palate. Bicester: Winslow Press. Watson, A.C.H., Sell, D.A. & Grunwell, P. (2001). Management of Cleft Lip and Palate, Chapters 14 snf 17. London: Whurr Publishers.

Bibliography Kelly, Alex. (2011). Talkabout for Children 1 – Developing Self Awareness and Self Esteem; Talkabout for Children 2 – Developing Social Skills. UK: Speechmark. Kelman, E. & Nicholas, A. (2008). Practical Intervention for Early Childhood Stammering – The Michael Palin Centre Approach. Milton Keynes: Speechmark. Meyers, S. & Woodford, L. (1992). The Fluency Development System for Young Children. NY: United Educational Services, Inc/DOK Publishers.

9 A pplying the Psycholinguistic Framework in Cleft Speech Therapy Benedicta Isaac-Kumar

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Aim of this chapter The aim of this chapter is to provide a brief overview of the psycholinguistic framework and its application to assessment and therapy with children with cleft palate. This chapter will explore how a child’s response to assessment stimuli can give us significantly more information once we apply psycholinguistic principles to it. In addition, it will illustrate how psycholinguistic principles can be used to help a child produce new sounds he has hitherto not attempted and therefore its relevance to therapy.

Psycholinguistics Psycholinguistics is the process of mapping a message a speaker intends to communicate onto its form (Goldrick, 2007). Psycholinguistic models therefore offer an approach to analysing speech as an output of language beyond the level of the acoustic signal. Levelt’s model of lexical access (1989) is the most well known theory, describing the process from conceptualisation to generation of a phonetic representation (spoken word) with specific articulatory gestures and timing. Treiman et al. (2003) emphasise the importance of the ‘mental lexicon’ where speech production represents the retrieval of words from this lexicon, along with their syntactic (grammar) and morphological (units of meaning) properties, a view also supported by Altmann (2001).

The psycholinguistic framework Psycholinguistic theory offers a connectionist perspective of the speech mechanism as a whole, from perception to production. The work of Stackhouse and Wells (2002, 1997) is perhaps most influential in speech and language therapy. Their Psycholinguistic Framework consists of a speech processing model mapped onto the speech chain from ear to mouth (Figure 9.1). It allows for consideration of the speaker’s stored ‘lexical representations’ of sounds and sound properties and the possibility that these can be replaced with different representations. The psycholinguistic framework is, for some, a mystery of boxes, arrows and lines. However, in dispelling these clouds of mystery, we discover a system that explains the theory behind many speech therapy activities we already

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Figure 9.1 Simplified Speech Processing Model – based on Stackhouse & Wells (1997)

use. Understanding the principles will also help us design some novel therapy activities.

Before we begin You may find it useful to access a copy of the original speech processing model (Stackhouse & Wells, 1997) and make notes on it as you go through this chapter. Remember that the system is complex and dynamic. The ‘boxes’ or stages are not watertight entities. Think ‘fluid and flowing’ as you work your way through it. Finally, the stages at the top are not more complex or important than the ones near the bottom. g Further Reading: Stackhouse, J. and Wells, B. (2007). Children’s Speech and Literacy Difficulties: A Psycholinguistic Framework. London: Whurr Publishers.

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Applying the psycholinguistic framework in assessment: Bella’s story Bella is 3 years old. She has a repaired bilateral cleft lip and palate. Her mother reports that her speech is unclear. I decide to administer the DEAP (Diagnostic Evaluation of Articulation and Phonology) to obtain a phonetic inventory and begin with the articulation assessment.

Target word: Door I take Bella through the picture-naming task. One of the items is ‘door’. Bella looks at the picture and confidently says [ɡɔ]. On seeing the picture and saying [ɡɔ], Bella has accessed her semantic representation – her understanding of the meaning of the word ‘door’ (i.e., that thing you open to go into your house). To produce the word, Bella then accesses her motor programme, which is the series of articulatory gestures needed to produce the word. Next is motor planning where the articulatory gestures are sequenced in real time. Finally motor execution, at the level of the mouth, where the articulators move to produce an acoustic signal for the word. Bella has therefore used a top-down processing strategy (Figure 9.2); starting with a skill at the top – semantic representation – then activating motor components lower down.

Why /ɡɔ/ for /dɔ/? Let’s assume that Bella knows that /ɡɔ/ and /dɔ/ are semantically different, i.e., one has a distinct semantic representation and the other does not, it is a nonsense word. Her errors could arise from breakdown at the following levels.

Breakdown at the phonological representation level Phonological representation is the basis for speech recognition. It contains just enough information of the word to distinguish it from other words known to the child.

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Figure 9.2 Top-down processing

Therefore phonological representations will have enough information to tell /pɪn/ from /fɪn/ but not /pɪn/ from /pʰɪn/ as this is not a phonological contrast. It may be that Bella recognises /ɡɔ/ and /dɔ/ as variations of the same word because the sounds /g/ and /d/ do not have different phonological representations. It is also possible that Bella hears the difference when an adult produces /ɡɔ/ and /dɔ/ but when she says them, her perception of the contrast is weak and the distinction not clear. A defective phonological representation will lead to a flawed motor programme, as motor programmes arise from the specifications stored in phonological representations. This is important to remember because output (speech) errors should not be viewed as just that; we could potentially be ignoring the root cause of the error, i.e., an input deficit.

Does the problem lie at the level of motor execution? This is the level where articulation is actually executed giving rise to an acoustic

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signal in the form of speech. We know that children with cleft palate often have difficulty producing alveolar sounds. Scarred palates providing little sensory feedback, absence of alveolar ridge (an important landmark in speech production), presence of anterior fistulae or some degree of VPD could all explain why Bella is ‘backing’ /d/ to [g].

Does Bella have difficulty accessing her motor programmes and are they accurate? This is harder to establish as the development of motor programmes is influenced by skills at other levels. If it is clear from assessment that Bella has accurate phonological representations, and if there is nothing to suggest difficulties at the level of motor execution, then it is possible the difficulty lies with inaccurate motor programmes. Also worth checking is Bella’s peripheral auditory processing (i.e., hearing) as children with cleft have a heightened risk of hearing loss, impacting on input skills.

Target word: Foot Let’s now move to another word from the DEAP assessment: ‘foot’. Seeing the picture stimulus, Bella says [kəʊz] for /təʊz/. I try offering a semantic cue

Figure 9.3 Bottom-up processing

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(‘you wear your socks on your … ’) to no avail. I then ask her to repeat the word after me and Bella responds by saying [fʊk]. One would expect ‘foot’ to be a word in Bella’s receptive vocabulary, but one for which a motor programme may not been established yet (i.e., not well rehearsed expressively). On hearing my model, Bella will have used her ‘motor programming’ facility to help her assemble the word. “Motor programming can be thought of as comprising a store from which phonological units are selected and a process of assembling these units in new combinations. An analogy would be a child’s box of letters from which you can make a selection, then sequence them in new combinations to create new words” (Stackhouse and Wells, 1997, p.163) Motor planning (and the units available in it) is dependent on the motor programme. This might explain why Bella, even when only imitating an adult model, uses a /k/ in place of a /t/.

Novel words vs. known words I now move on to the speech sound stimulability sub-test. Many words on this sub-test would be unknown to a child of three years of age (e.g., nigh, guy) and some words are nonsense words (e.g., ‘ing’). There are no picture stimuli used for this assessment and it is simply a repetition task.

Target word: Nigh One of the words I ask her to repeat is /naɪ/. Bella will most likely have no lexical representation for this. Lexical representation refers to all stored knowledge about the word and amongst other things will include semantic representation, phonological representation and motor programming. Without any ‘top’ information available, Bella cannot use a ‘top-down’ processing strategy on this task. She will therefore need a ‘bottom-up’ strategy, using phonological recognition and assembling a new motor programme. You can see how this repetition task has skirted around all pre-established knowledge. This is probably a good time to introduce the concept of ‘frozen motor programmes’ (Bryan & Howard, 1992). If a child has always said ‘tea’

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for ‘key’ (a normal early developmental error), with maturity, he acquires a /k/ which he uses in most new words (like ‘cough’ and ‘careful’), but /ki/ remains [ti]. This child is said to have a frozen motor programme, where some existing stored words have not been updated. Using words that will trigger bottom-up processing avoids stored words and knowledge and might be one way to help the child break from frozen motor programmes. Continuing with the DEAP stimuli list, I now ask Bella to repeat the word /θaɪ/. This is again probably a novel word for Bella with no stored knowledge and so is going to need a bottom-up strategy. I model /θaɪ/ with exaggerated tongue tip extension. Bella will most likely match this visual information to pre-existing units in phonological recognition and motor programming. She might decide from this that she is being asked to produce a brand new sound and will therefore create a new phonological unit in her phonological recognition store, leading to the creation of a brand new motor programme. The aim of this brief overview has been to get SLTs thinking that testing for psycholinguistic skills is not necessarily a whole new assessment, but rather interpreting available information, in the context of the psycholinguistic framework. An understanding of the underlying principles can help SLTs to target the right area of need during therapy by identifying the level at which the speech production system breaks down. Ignoring difficulties with input could, for example, lengthen the period of therapy, testing the child’s and therapist’s motivation.

Applying psycholinguistic principles in therapy: Timothy’s story The following case study will demonstrate the principle of using novel sounds in therapy, employing a bottom-up approach to eliminate entrenched speech sound patterns.

• Case study: Timothy

Timothy is 6 years old and presents with an active nasal fricative. He uses his active nasal fricative as a substitution for /s/. To create a sound that is auditorily and visually novel, we used interdental /s/. We also introduced ‘snorty-sauraus’, a dinosaur that wanted to be able to say his words but could only snort them.

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We started by devising a symbol for the active nasal fricative, in this case ‘5’. As Timothy was already reading, we devised a to represent an oral /s/. To make the distinction even more salient, we described the active nasal fricative as the ‘pointy-nose sound’ and the oral /s/ as the ‘curly tongue sound’. Timothy had lots of practice hearing the two sounds and deciding which one the dinosaur would like to use. We then swapped and Timothy had practice saying the two sounds while I decided which one was the better sound for the dinosaur. We used the symbols we had designed through every stage of therapy. We then practised short nonsense words /is, us, his/. We sometimes used ‘5’ in the word and sometimes , all the time judging which one the dinosaur should use more of, before moving on to real word targets. Timothy had lots of practice being the dinosaur and sometimes playing the therapist that helped the dinosaur. With targeted therapy and lots of input from his family, Timothy mastered the concept easily. The fact that he had no other phonological deficits or articulatory deficits helped him use and generalise the new sound fairly quickly.

Conclusion The aim of this chapter has been to give you a taste of psycholinguistics and its principles, using Bella and Timothy to demonstrate some aspects and applications of the principles to assessment and therapy. Not all children are textbook examples. There will always be those children with more challenging needs and baffling responses to stimuli. However, a thorough understanding of psycholinguistic principles will help you devise sound therapy activities and manipulate existing activities to plan achievable goals.

References Altman, G.T.M. (2001). The language machine: Psycholinguistics in review. British Journal of Psychology, 92, 129–170. Bryan, A. & Howard, D. (1992). Frozen phonology thawed: The analysis and remediation of a developmental disorder of lexical phonology. European Journal of Disorders of Communication 27, 343–365. Goldrick, M. (2007). Connectionist principles in theories of speech production. In M.G.

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Gaskell (Ed.), The Oxford Handbook of Psycholinguistics (pp.515–530). Oxford: Oxford University Press. Levelt, W.J.M. (1989). Speaking: From Intention to Articulation. Cambridge: MIT Press. Levelt, W.J. (1993). Lexical access in speech production. In E. Reuland & W. Abraham (Eds), Knowledge and Language: From Orwell’s Problem to Plato’s Problem, Volume I (p.241). NY: Springer. Stackhouse, J., Wells, B. & Snowling, M.J. (1997). Children’s Speech and Literacy Difficulties: A Psycholinguistic Framework. London: Whurr Publishers. Stackhouse, J., Wells, B., Pascoe, M. & Rees, R. (2002). From phonological therapy to phonological awareness. Seminars in Speech and Language, 23(1), 23–34. Stackhouse, J. & Wells, B. (2007). Children’s Speech and Literacy Difficulties. A Psycholinguistic Framework. London: Whurr Publishers. Treiman, R., Clifton, C. Jr., Meyer, A.S. & Wurm, L.H. (2003). Language comprehension and production. In Comprehensive Handbook of Psychology, Vol. 4; Experimental Psychology (pp.527–548.). NY: John Wiley & Sons, Inc.

10 Section 1 Oral Motor Therapy: Is there a place for it in speech therapy for children with cleft palate? Benedicta Isaac-Kumar

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Aim of this section The aim of this section is to gain some understanding on who might benefit from Oral Motor Therapy (OMT), particularly within the cleft population. The section takes a question-and-answer format and covers the rationale, goals and clinical applications of OMT, and the debate surrounding it.

Introduction The efficacy of OMT has been debated in the speech therapy world for some years (Lof, 2006; Williams et al., 2006; Clark, 2005; Forrest, 2002). This chapter is based on the premise that in this area ‘lack of evidence’ does not necessarily mean ‘no evidence’. It is also acknowledged that brain and neural networks remain poorly understood and the speech mechanism and its intricacies are still being elucidated.

OMT: How did it all begin? Marshalla (2008) traces the first use of the term ‘oral motor’ to Wilson (1978), referring to speech and feeding in children with neurological impairment. However, even prior to this publication, the literature is replete with authors, academicians and researchers advocating oral motor techniques. Marshalla’s review (2008) spanned the years 1912 through 2007 and found references to activities that, using current terminology, would be classified as oral motor therapies, starting with ‘tongue gymnastics’ recommended by Scripture in 1912. Therefore, while the term ‘oral motor therapy’ is a recent innovation, the concept itself spans many years of work beginning at least at the early part of this century. t Useful Link: The website, Oral Motor Institute, has brought together some excellent resources, explaining the history of oral motor therapies. http://www.oralmotorinstitute.org/

g Further Reading: Marshalla, P. (2012). Horns, whistles, bite blocks, and straws: A review of tools/objects used in articulation therapy by Van Riper and other traditional therapists. Oral Motor Institute, 4(2). Available at: http://www.oralmotorinstitute.org/mons/v4n2_marshalla.html

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Question: What exactly is OMT? An accurate description/definition of OMT is important. In fact, a review of the literature suggests that discrepancies in definitions have partly contributed to the debate and controversy surrounding OMT. “Oral Motor therapy…can be defined as the process of facilitating oral (lip, jaw, tongue) movements” Marshalla (2004), p.10 “…teaches oral structural placement to clients who cannot produce or imitate speech sounds using traditional auditory or visual input…Traditional speech facilitation has not been effective with these clients because there may be a movement or placement disorder” Rosenfeld-Johnson (2009), p.1 “Oral motor treatment addresses sensory processing as well as dissociation, grading, direction, timing, and coordination of mouth movement for eating, drinking, speaking, and other mouth activities” (2008a), poster; American Speech-Language-Hearing Association, Chicago, Illinois Drawing these definitions together, we arrive at an approach that: • Should have a clear feeding or speech goal • Should be used to treat a very specific population, i.e., clients with a movement or oral placement disorder • Involves oral activities (therefore any of the articulators) and motor skills, (therefore movement, sensation and placement).

• Terminology

Motor pertains to or involves muscular movement; movement is to some extent based on sensation and accurate movement and sensation creates accurate placement, thereby inextricably connecting movement, sensation and placement.

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• Note

Specific OMT approaches include: • Oral Placement Therapy (TalkTools®) – teaches oral structural placement for speech (Rosenfield-Johnson, 2009) • PROMPT (Prompts for Restructuring Oral Muscular Phonetic Targets) – uses tactile cues to help the child shape articulation (Grigos et al., 2010) The Nuffield Dyspraxia Programme is not an OPT approach but includes work on speech motor skills (Williams & Stephens, 2004).

Question: What is not OMT? It is difficult to clearly separate OMT approaches from Non-Speech Oral Motor Exercises (NSOME), as illustrated by the definition of NSOME from Lof (2008) below: “any technique that does not require the child to produce a speech sound but is used to influence the development of speaking abilities” Lof and Watson (2008), p.394 As they develop this subject, Lof and Watson (2008), go on to criticise NSOME as being ‘decontextualised’ techniques that ‘disintegrate the highly integrated task of speaking’. Lass and Pannbacker (2008) use the term Non-Speech Oral Motor Treatments (NSOMT) to describe non-speech movements of the speech mechanism, e.g., exercises, blowing, icing and swallowing. ‘Wagging the tongue’ has often been quoted as an example of an NSOMT that has no relationship to real speech. However, no author who has recommended such an activity has even remotely implied it would be the sole path to speech clarity. Instead, non-specific tasks such as this were recommended for three basic reasons (Marshalla, 2008): 1. To facilitate awareness of the oral mechanism. 2. To intensify or ‘vivify’ oral movement.

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3. To achieve some other neuromuscular end, e.g., to increase tone, range of movement, integration or differentiation of oral movements. It is inevitable then that OMT will include NSOME/NSOMT but without a clear point of transition from simply ‘oral motor’ to speech itself, NSOME remain just that. While blowing and puffing up cheeks are oral motor activities, they will not qualify for classification as Oral Motor Therapy until they are used intending to treat speech or eating difficulties. Whether a task can be classified as NSOME or OMT therefore lies in the hands of the practising therapist. It depends on whether the oral motor task has been chosen with a clear rationale and whether its transition into a speech skill has been planned in advance. The key is to always remember that OMT is an approach and not a goal. It does not aim to strengthen muscles for the sake of stronger muscles; the rationale for use is the anticipation of clearer speech or improved feeding.

Question: What is the controversy? The controversy surrounding OMT leaves most therapists very unsure of its value, or otherwise, in clinical practice. Clark (2003) identifies the lack of evidence in this area and comments that aspects of neuromuscular function remain virtually unstudied in relation to speech and swallowing.

• Research

Two surveys developed by Bahr (2008/2009) were circulated amongst 500 speech therapists. The results showed that 32–74% reported hearing negative comments about oral motor therapy including ‘Oral motor treatment does not work’ and ‘There is no research on oral motor treatment’. However, a survey by Lof and Watson (2004, 2008) showed that 85% of SLTs used nonspeech oral motor exercises (NSOME) to treat speech sound productions and 61% of surveyed SLTs agreed with the statement: ‘The literature I have read strongly encourages the use of NSOME’. Is there then a group of therapists who are hearing conflicting information on OMT and are generally unsure about the efficacy of it, but are using it anyway? The approach that a therapist selects to treat a disorder will depend on the apparent deficits identified through assessment and the therapists’

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understanding of the theory behind the treatment. When faced with poorly researched, complex disorders such as dysarthria and dyspraxia, it is even more important to stay abreast of developments in theory and practice, as decisions about treatment are harder to make.

The argument against… Lof (2009) summarises the reasons why NSOME should not be used to treat speech sound disorders, by considering the following questions, which are also applicable to OMT: 1. Is strength necessary for speaking? Lof (2009) cites the evidence of Sudbery et al. (2006) that pre-school children with speech sound disorders may actually have stronger tongues than their typically developing peers. 2. How relevant are NSOME to speech? 3. Lof argues that the context in which a skill is learned is crucial. Therefore it inappropriate to practise tongue elevation to the alveolar ridge with the desire that the isolated task will improve production of the lingual-alveolar sound /s/. 4. Can young children take advantage of the non-speech mouth cues provided during NSOME and transfer these to speaking tasks? Lof argues that although the same structures are used for speaking and other mouth tasks they function in different ways depending on the task and each task is mediated by different parts of the brain. This should spell the death knell for our beloved Mr Tongue! What should we do then? Should we instantly abandon all approaches that do not currently meet the standards of evidence-based practice? The following quote from Clark (2003) provides us with some food for thought: “At least two strategies are available to clinicians selecting management techniques for specific individuals: The approach that is advocated by evidence-based practice is to refer to research reports describing the benefits of a particular treatment. The question asked in this case is, ‘Is this treatment beneficial?’ In the absence of adequately documented clinical efficacy, clinicians may select treatments based on theoretical

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soundness. The question asked in this case is ‘Should this treatment be beneficial?’ This second method of treatment selection has potential for success if the clinician has a clear understanding of both the nature of the targeted impairment and the therapeutic mechanism of the selected treatment technique” Clark (2003), p.400

The argument for… Therapists who believe there is a place for OMT in speech therapy are basing it on the premise that speech is a muscular activity and therefore any abnormality within the muscular system will likely disrupt speech. The rationale for OMT is therefore that treating the aberrant motor process will improve speech function. An evidence-based systematic review by McCauley et al. (2009) concluded that there was insufficient evidence to support or refute the use of oral motor exercises to produce effects on speech. In conclusion, therefore, OMT is neither obsolete nor new-fangled, but it is clearly lacking in evidence. Whilst opposition to the approach remains strong, the needs of children with speech disorders remain and it could be argued that OMT, for them, is an option worthy of consideration and further systematic evaluation.

Question: What specific activities does OMT include? The choice and range of OMT activities is as extensive as its history. Marshalla (2008) provides a comprehensive review, from various authors, described over a number of years. Examples are given below: • Tactile cues: e.g., pressure applied under the child’s chin, upward and toward the base of the tongue, to reinforce back-of-tongue productions (Blakely, 1983, p.30) • Assisted oral movements: e.g., pull down the chin with your index finger for production of /a/ (Kaufman, 2006, p.14) • Describing oral movements: e.g., to describe tongue position for /t/, discuss how this sound is produced, including how the tongue is positioned on the alveolar ridge (Folk, 1992, p.6)

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• Developing general oral sensory awareness: e.g., chew in an exaggerated fashion or practise licking the lips and cleaning the tongue and cheeks with the tongue (Van Riper, 1954, pp.216–218) • Maintaining oral movements: e.g., to maintain lip-to-lip articulation for swallowing, the patient may be asked to hold lip closure for one minute, repeated 10 times per day (Logemann, 1983, pp.145–146) • Practising oral movements: e.g., for parents to practise lip movements with their children, blow bubbles, blow kisses, blow whistles, hum your favourite song together (Dougherty, 2005, p.89) • Stabilising oral movements: e.g., to stabilise the lips and facial muscles with low muscle tone, play pat-a-cake, peek-a-boo, and other children’s games that incorporate patting, tapping and stroking of the cheeks and lips (Morris & Klein, 2000, p.445) • Stimulating reflexive oral movements: e.g., to stimulate elevation of the back of the tongue for velar sounds, back elevation can be facilitated by stimulating the Tongue Retraction Response (TRR). Stroke down the middle of the tongue to about half way toward the back, the whole tongue humps up and back into a ball shape which completely fills the posterior oral cavity and occludes the airway (Marshalla, 1992, p.98). The list above highlights the range of OMT activities. While some fall more obviously within the description of oral motor tasks, such as the popular ‘Mr Tongue’, clinicians may use others in practice, such as tactile cues, without thinking of them specifically as oral motor. Others may be perceived as being too invasive to be used within a community clinical setting. This last point may be another reason why OMT has fallen out of fashion: health, safety and hygiene regulations may, if only subconsciously, discourage physical contact in therapy.

Question: Who could benefit from OMT? Speech is a motor skill. In principle, therefore, OMT is for children with ‘speech’ difficulties. Speech can be defined as a fine motor skill because:

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1. It is performed with accuracy and speed

2. It uses knowledge of results 3. It is improved by practice 4. It demonstrates motor flexibility in achieving goals 5. It relegates all of this to automatic control. (Netsell, 1982)

Explaining this in terms of disorder type, OMT is likely to benefit children with articulation difficulties/disorder. The ongoing controversy surrounding OMT may in part be accounted for by confusion between what constitutes a phonological difficulty/disorder and speech errors that are articulatory in origin. For example, use of the term ‘speech sound disorders’ in relation to the effectiveness or otherwise of OMT is not helpful as this could refer to children with speech difficulties of phonological and/or articulatory origin. As a case in point, Lof (2006) suggests that OMT should not be used with children with speech sound disorders, quoting Velleman and Viheman (2002) who recommend the use of ‘Whole-Word Phonology and Templates’. Let’s consider a hypothetical child who has arrived for speech therapy presenting with consonant deletion in connected speech. The therapist typically begins by administering an assessment which shows that the child has acquired the full range of English sounds used consistently in small words. The child has no difficulty with discriminating between sounds and testing indicates there are no further psycholinguistic deficits. Where should therapy begin? It appears there are no obvious therapy targets, but there are definite concerns about speech clarity. Closer examination shows difficulties of another nature. The child sometimes speaks on an ingressive airstream (though this is not easily noticeable). She is not able to raise her tongue independent of her jaw. She cannot puff up her cheeks. There is a history of poor coordination whilst feeding, in addition to mild gross motor coordination difficulties. Children with difficulties of this nature often slip through the net, as they compensate for difficulties without any direct input. This is also the child that therapists are often tempted to discharge with written advice to school and family on ‘how to support a child with unclear speech’.

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Question: How does the use of ‘Tools’ fit into OMT? The use of ‘tools’ could be considered an essential part of speech therapy, with flexibility on the therapists’ part to use and adapt whatever is available. Marshalla (2012) reviewed 14 speech therapy textbooks between 1939 and 1968 and found 86 different objects or types of object that were recommended for use in articulation therapy. The list includes bubbles, balloons, cotton, matches, mirrors, pencils, probes, tongue depressors, recorders and tooth picks. Some of these tools were hard to use, some had not been health and safety tested and some only just about did the job. Many, of course, are widely available and not custom-made for articulation therapy or OMT. There is evidently some antipathy towards designing, selling or recommending such custom-made tools in speech therapy: “OMT is in your face. Trade displayers at conferences adorn their bosoms with cute rubbery pendants; catalogues and professional publications glisten with advertisements for Ps and Qs; straws, horns, chewy tubes, bubbles, bite blocks mean big business; special offers and freebies abound” (Bowen, 2005, p.145) There is no doubt that marketing claims must be viewed cautiously. However, if a child arrives at clinic and has limited tongue tip movement (and consequently a tongue blade /t/), where are we going to begin? Are we going to endlessly model /t/ and work on discrimination when there is no evidence that this particular child needs this, frustrating ourselves and the child, who does not respond to the ‘say after me’ approach to therapy? Or would it be worthwhile considering an OMT approach, with or without custom-made tools? The following is a case study of a child who benefitted from an OMT approach, using custom-made (Talk-Tools®) and other tools.

• Case study: Joseph

Joseph was first referred to the cleft team, aged 4 years. He had non-cleft VPD and, following a sphincter pharyngoplasty, had a competent velopharyngeal mechanism. He presented with backing of alveolar sounds, but interestingly, as he spoke, there were quite asymmetric tongue movements. He appeared to be a generally hypotonic child, with a history of feeding difficulties and was under the care of a paediatrician.

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Assessment While the tongue appeared to be the source of Joseph’s difficulties, other articulatory mechanisms were assessed and any difficulties addressed. The jaws form the framework within which speech production occurs. Following assessment, it was established that Joseph had unstable asymmetric jaw movements. Joseph therefore initially worked through a series of bite blocks, which included activities such as biting and biting with resistance for increasing lengths of time. Breathing and phonation: Joseph then worked through a series of activities, designed to improve breath control. Joseph had been described by his parents as running out of breath easily and tiring quickly. Bubble blowing exercises and blowing recorders arranged in an order of increasing difficulty were used to address this. Tongue: Next to be addressed were the abnormal tongue movements. One recommended TalkTools® activity is a therapeutic straw exercise: a series of eight straws, arranged in order of difficulty, each addressing a specific goal such as improving tongue retraction, improving jaw stability and improving jaw-tongue-lip dissociation (the ability to move lips or tongue independent of the jaws). This was followed by specific tongue tip activities. Joseph was asked to poke his tongue out and, using a highlyflavoured liquorice stick, stimulate his tongue in three rapid posterior to anterior strokes. This helped to produce a neat tongue tip which could then be immediately transitioned to a tongue tip activity. The tongue tip activity in this case was the Cheerio™ exercise (see below). Following this programme, Joseph was able to produce a /t/ in isolation. Once the sound was established in isolation, Joseph worked on blending the sound with vowels and is currently learning to use the sound in short words ending with /t/.

• Note:

The Cheerio™ exercise involves placing one of the cereal hoops on the child’s alveolar ridge, instructing him to place his tongue in the hole and holding it there for 5 seconds. When the child can do this consistently 50 times he is ready to transition to alveolar sounds and then move on to traditional articulation therapy. (Please note this is only a truncated version of the original exercise which can be found in: Oral Placement Therapy for Speech Clarity and Feeding, by Sarah Johnson-Rosenfeld (2009).)

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Question: Can children with cleft lip and palate benefit from OMT? A child with a cleft lip and palate is not necessarily lacking in tongue or lip strength, although in some cases a child may have associated difficulties, including dyspraxia or dysarthria. While children with cleft lip and palate do not necessarily need input to increase strength or range of movements, placement accuracy might be hard to achieve, as shown from the example below.

• Case study: Max

Max is 4 years old with a repaired bilateral cleft lip and palate. His repaired palate has no alveolus; the palate is very narrow with some supernumerary teeth at the site of the alveolus. He also has an anterior fistula, due to be closed at the time of his alveolar bone graft (therefore not until he reaches 7–11 years). Max instinctively avoided the alveolar ridge as a place of articulation for the above reasons and therefore backed alveolar sounds to velar. The Cheerio™ task (described above) gave him a good idea of tongue placement, which could be gently transitioned to whispered alveolar sounds from where traditional articulation therapy took over. As Max only had sensory/placement inaccuracies, we did not need to do any further OMT activities. Further therapy targeted phonological contrasts, as this was also identified as a weak area for Max. More commonly, children with cleft or non-cleft-related VPD or with diagnosed syndromes and associated cleft present with complex speech arising from the coexisting conditions (for example, a combination of compensatory articulation found together with dyspraxia or VPD together with dysarthria). Children with 22q11 deletion syndrome are such a group in whom complexities of speech disorder are commonly noted (Kummer et al., 2007; Scherer et al., 1999; Golding-Kushner, 1995). It is with these complex children that OMT may be considered as an option. Remember, however, that a child with compensatory glottal articulation will show limited lip and tongue movements. This child does not necessarily have either dyspraxia or dysarthria. In the presence of VPD he/she has valved air flow at the level of the glottis to produce his/her compensatory consonant sounds (glottals) and therefore does not need to execute any further articulatory movements using the tongue or lips.

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• Case study: Joshua

Seven-year-old Joshua was referred with a diagnosis of CHARGE syndrome. In keeping with his diagnosis, he had abnormalities of cranial nerve VII, causing unilateral facial palsy. When he was first referred he also had noncleft velopharyngeal dysfunction (VPD). Joshua’s facial palsy caused an open mouth posture with a mild tongue thrust over which he seemed to have little control. Joshua had unintelligible speech with only non-oral sounds (a mixture of glottal, pharyngeal and nasal). Perhaps not surprisingly, Joshua was also a very messy eater. CHARGE syndrome

CHARGE is a complex medical birth condition where the baby is born with heart abnormalities and breathing difficulties. Children also typically have hearing loss, vision loss and balance problems. CHARGE was an acronym originally coined in the 1980s to describe this condition where C stands for coloboma of the eye, H for heart defects, A for atresia of the chonae, R for retardation of growth and/or development, G for genital and/or urinary abnormalities and E for ear/hearing abnormalities. Although the condition is no longer diagnosed by the above features, the name remains unchanged. Intervention: One of Joshua’s early targets was production of /w/, which was rather hard to achieve. At that point, Joshua had had his first palate surgery (palatoplasty) and it was suspected he still had an incompetent palate. Whilst waiting to gather enough evidence for further surgery, low pressure consonants seemed a reasonable target. However, Joshua’s very low lip strength precluded the smooth lip glide needed for production of /w/. Joshua worked through bubble blowing activities and a series of recorders, designed to improve lip closure and lip rounding. This helped elicit round lips (which Joshua could previously not do). Round lips accompanied by voicing produced /u/, then /w/ was taught as a transition from /u/ to a vowel. (Try it yourself: say a long /u/ and then transition smoothly into another vowel, /i/ for example and you will hear a ‘wee’.)

Note When using any blowing exercises with a child with VPD, establish that the child has sufficient palatal competence to carry it out. Forceful blowing in the presence of VPD could lead to ‘tongue humping’, where the child uses the back of their tongue to assist palate closure, possibly encouraging

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a backing process. Nose-holding is therefore recommended to assist gentle blowing in the presence of VPD (see Chapter 7). Another early goal was production of /p/. With little ability to consistently close his lips, building air pressure behind them and releasing it to produce a /p/ was not a reasonable expectation of Joshua. OMT helped Joshua start his speech journey at a more appropriate place. His new goal, therefore, was to improve lip closure for improved production of bilabial speech sounds. Some eating and drinking goals were also incorporated, which mirrored his speech goals, initially targeting better lip closure, decreasing tongue thrust and improving jaw stability. Progress: Joshua worked through a series of straws and recorders, arranged in an order of increasing difficulty. Joshua’s increasing control over his lips could now be harnessed for sound production. Again following specific transition activities (in this case, using a tongue depressor held between his lips to provide increased sensory feedback, and saying the sound around the depressor), his attempts at /p/ were now more consistent and accurate. Joshua’s tongue movements were targeted next. Alongside this were eating/drinking goals, which paralleled his speech production goals. The targets were to improve tongue lateralisation and tongue tip elevation. Joshua was now moving satisfactorily from his open mouth, tongue thrust posture to using a more natural tongue posture at rest and showing more control over tongue lateralisation and tongue tip elevation. The use of specific tasks for transferring the newly acquired oral motor skill into a speech skill helped achieve production of /t/ (specifically, the Cheerio™ task, details of which were discussed earlier in this chapter). This was followed by traditional articulatory work to include the sound into vowel-consonant (VC) combinations, consonant-vowel (CV) combinations and so on. Summary: Joshua continues to receive speech therapy. Using OMT has helped prepare him for traditional therapy and acquire a range of sounds in isolation. The bulk of his work is now traditional articulation therapy and generalisation activities. Joshua’s experience points to a child with whom NSOME/NSOMT activities were used as part of an OMT approach, with a clear point of transition into speech production and a clear end point: more intelligible speech and improved eating and drinking.

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Conclusion This section has considered whether there is a place for oral motor therapy in speech therapy for children with cleft palate. This includes approaches that are described as using oral motor or oral placement activities and may also include non-speech oral motor exercises; although a clear transition to speech or feeding tasks must be evident. The ongoing controversy surrounding oral motor therapy is acknowledged along with the inadequacy of the existing evidence base. Several case studies illustrate how the approach has been usefully included as part of a wider speech and language therapy programme for children with a cleft palate and demonstrate the need for further systematic evaluation of the efficacy of the interventions.

References Bahr, D. (2008, November). The oral motor debate: Where do we go from here? Poster session presented at the annual meeting of the American Speech-Language-Hearing Association, Chicago, IL (Full handout available from http://convention.asha.org/handouts/1420_2054 Bahr_Diane_124883_Nov03_2008_Time_103047AM.doc). Bahr, D. (2011). The oral motor debate part II: Exploring terminology and practice patterns. Oral Motor Institute, 3(2). Available from www.oralmotorinstitute.org. Blakely, R.W. (1983). Treatment of developmental apraxia of speech. In W.H. Perkins (Ed.), Current Trends of Communicative Disorders: Dysarthria and Apraxia (pp.25–34). New York: Thieme. Clark, H.M. (2003). Neuromuscular treatments for speech and swallowing. American Journal of Speech-Language Pathology, 12, 4; Pro Quest Psychology Journals, 400–415. Clark, H.M. (2005, June 14). Clinical decision making and oral motor treatments. The ASHA Leader. www.asha.org. Dougherty, D.P. (2005). Teach Me How to Say it Right. Oakland: New Harbinger. Folk, M.J. (1992). Straight Speech. Vero Beech, FL: Speech Bin. Forrest, K. (2002). Are oral-motor exercises useful in treatment of phonological/articulation disorders? Seminars in Speech and Language, 23, 15–25. Golding-Kushner, K.J. (1995). Treatment of articulation and resonance disorders associated with cleft palate and VPI. In R.J. Shprintzen & J. Bardach (Eds), Cleft Palate Speech Management: A Multidisciplinary Approach, (pp.327­–351). St. Louis: Elsevier Mosby. Grigos, M.I., Hayden, D. & Eigen, J. (2010). Perceptual and articulatory changes in speech production following PROMPT treatment. Journal of Medical Speech & Language Patholology, 18(4), 46–53.

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Kaufman, N. (2006). The Kaufman Speech Praxis Workout Book: Treatment Materials and Home Program for Childhood Apraxia of Speech. Gaylord, MI: Northern Rehabilitation Services. Kummer, A.W., Lee, L., Stutz, L.S., Maroney, A. & Brandt, J.W. (2007). The prevalence of apraxia characteristics in patients with velocardiofacial syndrome as compared with other cleft populations. Cleft Palate-Craniofacial Journal, 44, 175–181. Lass, N.J. & Pannbacker, M. (2008,). The application of evidence-based practice to non-speech oral motor treatments. Language, Speech, and Hearing Services in Schools, 39, 408–421. Lof, G.L. (2002). Two comments on this assessment series. American Journal of SpeechLanguage Pathology, 11, 255–257. Lof, G.L. (2003). Oral motor exercises and treatment outcomes. Perspectives on Language, Learning and Education, 10(1), 7–12. Lof, G.L. (2004). What does the research report about non-speech oral motor exercises and the treatment of speech sound disorders? http://www.apraxia-kids.org/library/whatdoes-the-research-say-regarding-oral-motor-exercises-and-the-treatment-of-speechsound-disorders/ Lof, G.L. & Watson, M. (2004). Speech-language pathologist’s use of non-speech oral-motor drills: National survey results. Poster presented at the annual meeting of the American Speech-Language-Hearing Association, Philadelphia, PA. Lof, G.L. (2006). Logic, theory and evidence against the use of non-speech oral motor exercises to change speech sound productions. Invited presentation at 2006 ASHA Convention. Lof, G.L. (2008). Introduction to controversies about the use of non-speech oral motor exercises. Seminars in Speech and Language, 29(4), 253–256. Lof, G.L. & Watson, M. (2008). A nationwide survey of non-speech oral motor exercise use: Implications for evidence based practice. Language, Speech and Hearing Services in Schools, 39, 392–407. Lof, G.L. (2009). Non speech oral motor exercises: An update on the controversy. American Speech-Language-Hearing Association Convention, New Orleans, LA, November 20, 2009. Logeman, J.A. (1983). Evaluation and Treatment of Swallowing Disorders. San Diego: College-Hill. McCauley, R.J., Strand, E., Lof, G.L., Schooling, T. & Frymark, T. (2009). Evidence-based systematic review: Effects of non-speech oral motor exercises on speech. American Journal of Speech-Language Pathology, 18, 343–360. Marshalla, P.J. (1992). Oral Motor Techniques in Articulation and Phonological Therapy. Seattle: Innovative Concepts. Marshalla, P. (2004). Oral Motor Techniques in Articulation and Phonological Therapy. Mill Creek, WA: Marshalla Speech and Language. Marshalla, P. (2008). Oral motor treatment vs. non-speech oral motor exercises. Oral Motor Institute, 2(2). Available from www.oralmotorinstitute.org.

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Marshalla, P. (2012). Horns, whistles, bite blocks, and straws: A review of tools/objects used in articulation therapy by Van Riper and other traditional therapists. Oral Motor Institute, 4(2). Available from www.oralmotorinstitute.org. Morris, S.E. & Klein, M.D. (2000). Pre-Feeding Skills: A Comprehensive Resource for Mealtime Development, 2nd Edn. Austin: Pro-Ed. Netsell, R. (1982). Speech motor control and selected neurological disorders. In S. Grillner, B. Lindblom, J. Lubker & A. Persson (Eds), Speech Motor Control. Oxford: Pergamon Press. Rosenfeld-Johnson, S. (2009). Oral Placement Therapy for Speech Clarity and Feeding. Tucson, AZ: Talk Tools/Innovative Therapists. Scherer, N.J., D’Antonio, L.L. & Kalbfleisch, J.H. (1999). Early speech and language development in children with velocardiofacial syndrome. American Journal of Medical Genetics (Neuropsychiatric Genetics), 88(6), 714–723. Scripture, E.W. (1912). Stuttering and Lisping. New York: MacMillan. Sudbery, A., Wilson, E., Broaddus, T. & Potter, N. (2006, November). Tongue strength in preschool children: Measures, implications, and revelations. Poster presented at the annual meeting of the American Speech-Language-Hearing Association, Miami Beach, FL. Velleman, S. & Vihman, M. (2002). Whole-word phonology and templates: Trap, bootstrap or some of each? Language, Speech and Hearing Services in Schools, 33, 9–23. Williams, P. & Stephens, H. (Eds) (2004). Nuffield Centre Dyspraxia Programme. Windsor, UK: NCDP Ltd. Williams, P., Stephens, H. & Connery, V. (2006). What’s the evidence for oral motor therapy? Acquiring Knowledge in Speech, Language and Hearing, Speech Pathology Australia, 8, 89–90. Wilson, J.M. (Ed.) (1978). Oral-motor Function and Dysfunction in Children. Conference proceedings, May 25–28, 1977. Chapel Hill: University of North Carolina.

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Bibliography Bahr, D. (2008). A topical bibliography on oral motor assessment and treatment. Oral Motor Institute, 2(1). Available from www.oralmotorinstitute.org. Bahr, D. (2011). The oral motor debate Part I: Understanding the problem. Oral Motor Institute, 3(1). Available from: www.oralmotorinstitute.org. Bahr, D. & Banford, R.J. (2012). The oral motor debate Part III: Exploring research and training needs/ideas. Oral Motor Institute, 4(1). Available from www.oralmotorinstitute. org. Banotai, A. (2007). Reviewing the evidence: Gregory Lof ’s critical take on oral-motor therapy. Advance for Speech-Language Pathologists & Audiologists, September, 7–9. Baylis, A.L., Munson, B. & Moller, K.T. (2008). Factors affecting articulation skills in children with velocardiofacial syndrome and children with cleft palate or velopharyngeal dysfunction: A preliminary report. Cleft Palate Craniofacial Journal. 45 (2), 193–207. Bowen, C. (2005). What is the evidence for oral motor therapy? Acquiring Knowledge in Speech, Language and Hearing, 7, 144–147. Bunton, K. (2008). Different tasks, different neural organisation. Seminars in Speech and Language, 29(4), 267–275. Connecticut State Department of Education (2008). Guidelines for feeding and swallowing programmes in schools. http://www.sde.ct.gov/sde/lib/sde/PDF/DEPS/Special/Feeding_ and_Swallowing.pdf Hammer, D. (2007). Childhood apraxia of speech: New perspectives on assessment and treatment. Las Vegas, NV: The Childhood Apraxia of Speech Association (workshop). Hodge, M., Salonka, R. & Kollias, S. (2005, November). Use of non-speech oral-motor exercises in children’s speech therapy. San Diego, CA: Annual Meeting of the American Speech-Language-Hearing Association. Ingram, D. (1976). Phonological Disability in Children. London: Edward Arnold. Lof, G.L. & Watson, M. (2005, November). Survey of universities’ teaching: Oral motor exercises and other procedures. Poster presented at the Annual Meeting of the American Speech-Language-Hearing Association, San Diego, CA. Marshalla, P. (2007). Oral motor techniques are not new. Oral Motor Institute, 1(1). Available from www.oralmotorinstitute.org. Ruscello, D.M. (2008). Non-speech oral motor treatment issues related to children with developmental speech sound disorders. Language, Speech and Hearing Services in Schools, 39, 381–391. Secord, W.A., Boyce, S.E., Donohue, J.S., Fox, R.A. & Shine, R.E. (2007). Eliciting Sounds: Techniques and Strategies for Clinicians, 2nd Edn. Clifton Park: Thomson Delmar Learning. Van Riper, C. (1954). Speech Correction: Principles and Methods, 3rd Edn. New York: Prentice-Hall.

10 Section 2 Active Nasal Fricative (ANF) Helen Piggott and Fiona Jeyes

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Aim of this section • To describe the nature of ANF. • To describe how the clinician can confidently diagnose ANF. • To distinguish ANF from speech sound substitutions resulting from VPD. • To suggest ideas for successful therapy approaches, plans and resources.

Introduction Although often heard in the context of VPD, ANF can occur in the speech patterns of those with a normal velopharyngeal mechanism. In such cases where there is velopharyngeal mislearning the speaker is using a substitution described as active nasal fricative, which perceptually presents as either nasal turbulence or emission replacing the target sound (Harding & Grunwell, 1998). This can give speech an overall hypernasal quality and raise concerns about palate function for speech. Clinicians should be aware that children referred with ‘nasal’ speech and queries regarding their VP function may present with this type of sound-specific nasality, which does not necessarily indicate that there is any issue with palate function.

What is an active nasal fricative? This is a sound which is heard as a substitution for a range of plosives/pressure consonants: /f/ /s/ /z/ /ʃ / /ʧ/ /ʤ/ can all be affected, although most frequently ANF replaces /s/ and /z/. ANFs are seen in children with cleft palate and VPD, as a compensatory articulation, but are also evident in children with no palate problems. In all situations they are mislearned sounds and are corrected by speech and language therapy, and not surgery.

What does an active nasal fricative sound like? It is perceived as a voiceless nasal fricative sound produced by directing air nasally rather than orally. The sound can be quiet or turbulent, and is sometimes described in the literature as ‘nasal snort’ (Golding-Kushner, 2001).

Active Nasal Fricative (ANF)

ANF with nasal emission m̥͋

n̥͋

ŋ̥͋

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ANF with turbulence m̥̃̃

ñ̥̃

ŋ̥̃̃

How is ANF produced? An articulatory stop is made within the oral cavity. This can be bilabial, alveolar or velar. The soft palate is lowered and air actively directed through the nose as shown in Figure 10.2.1. It is important to realise, therefore, that there will need to be an articulatory as well as manner change before the sound is corrected.

How can ANF be diagnosed? First of all, listen to the child’s speech: are other consonant and vowel sounds clearly produced with no evidence of hypernasality? It can be helpful to listen to sentences that contain no fricative targets (e.g., ‘Daddy painted a door’, ‘Baby liked her bottle’, ‘Lola would not eat her cake’). Removing the fricatives from the sample will allow for easier listening to vowels without them being

Figure 10.2.1 Alveolar closure, lowered soft palate, air directed through the nose for ANF

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Figure 10.2.2 Nose-holding: air forced into Eustachian tube

contaminated by the nasal air emission. The impact on the production of adjoining vowel sounds when the palate is being lowered (to produce the active nasal fricative) should not be underestimated, especially when it is replacing high frequency sounds, e.g., /s/ and /z/. Nose-holding. Ask the child to say either an extended sound /sː/, or a phrase with the target sound repeated (e.g., ‘see saw see saw see saw’). While he/she is speaking place your fingers either side of his/her nose and gently occlude the nostrils as in Figure 10.2.2. What usually happens is that the sound stops suddenly and the child looks extremely uncomfortable. This is because the air has nowhere to go but up the Eustachian tubes and into the middle ear. Sometimes nose-holding will result in the sound being released as a plosive, e.g., /d/. Both outcomes are indicators of an active nasal substitution.

Differential diagnosis ANF replaces the target fricative completely rather than nasal emission, which accompanies target sounds, although this can be difficult to differentiate in

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connected speech. Occasionally, nose-holding facilitates production of the correct target fricative sound and this can be an indicator of VPD. Here, blocking the nose has mimicked velopharyngeal closure and in doing so has facilitated correct oral production of the sound. This is described as a passive nasal fricative. In this case, the entire speech sample should give an indication of problems with VP closure; in particular, there is likely to be evidence of hypernasality affecting vowels. It is generally considered that passive nasal sounds will not be corrected by speech therapy alone (Golding-Kushner, 2001; Watson, Sell & Grunwell, 2001).

Why might I hear ANF in children without cleft palate or VPD? There appears to be an association of ANF with children who have glue ear, or restricted airways due to enlarged adenoids/tonsils (Peterson-Falzone et al., 2001). It may seem counterintuitive that a child with a blocked nose would learn to push air through it, but it is so. It could be that in early development this is the easiest sort of friction that a child can achieve and that the incorrect pattern is then maintained (Hattee et al., 2001).

ENT considerations Children with ANF are sometimes referred to speech and language therapists by ENT surgeons who are cautious about removing adenoids when there is a query about palate function. Caution is appropriate as adenoidectomy will increase the effect of any underlying VPD and can lead to hypernasal speech. Adenoidectomy should be avoided if VPD is suspected and investigation of palatal function may be required. In relation to tonsils, however, there is clinical evidence that enlarged/ obstructive tonsils may interfere with palate function (Henningson & Isberg, 1988; Shprintzen et al., 1987). Therefore, where removal of tonsils is clinically indicated this may actually improve palate function and speech resonance.

Therapy for ANF Before embarking on therapy for ANF it is important to establish where the child is making the oral closure during sound production, as ANF can be part of a generalised backing pattern where a velar nasal is the substituted sound. The suggested therapy approach is based on a combination of the

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psycholinguistic principles discussed in Chapters 7 and 9 as well as a traditional articulatory approach. The psycholinguistic element is the use of a ‘novel sound’ in place of the target sound produced as an ANF. The sound used will be different from the target sound and will replace it until the child has established the target oral sound in his/her repertoire. The articulatory element involves modelling and describing the placement and manner of the novel sound and exploring how it can be shaped into the target sound. This therapy approach has been shown to be successful in a variety of clinical settings, but will need careful explanation to parents or to education staff who are carrying it out in practice. The idea of teaching a novel/adapted sound can sometimes feel wrong, especially to education staff, as they may fear that it contradicts the literacy programme which the child is learning. It is therefore important to ensure that parents and education staff understand how the ANF is produced and that it is not amenable to change with speech sound discrimination approaches alone. When approached in the right way, ANF can quite quickly be improved, in terms of producing the new oral sound. Generalisation, as with most new sounds, may take longer.

Introducing a novel sound using oral airstream: 1. Introduce the idea of a novel sound. Examples of these could be: ‘train slowing down’ [ts͡] ‘long ‘t’ [ts͡] ‘cola can opening’ [ts͡] ‘learner or baby snake’ [θ] ‘leaky balloon’ [ɸ] 2. Use a symbol/picture that is only associated with this sound. Sometimes the child will enjoy naming the novel sound and so has some ownership of the process of therapy. Each therapist will find the terminology that suits them and the stage of the child’s development. Different sounds and symbols work for different children; it is a good idea to try a range of sounds and then use whichever seems to work best for that child. If the child is backing their articulation, it might be better to use a sound with tongue protrusion to break this pattern.

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3. The symbols/pictures used should not be part of a reading

scheme which is already known to the child. Avoid using letters written on the symbol, as some children will already have learned to associate these with their ‘old’ sound. The novel sound is referred to as the child’s ‘new sound’. (See Resource CD for ideas for picture symbols.)

4. Spend some time practising the novel sound in isolation. Talk about how it is a ‘mouth’ sound. Feel the air coming out of the mouth. Show the child how they can blow a feather/bubble on a wand/silly soap blob, with the air coming through their lips. It may help to show that the sound comes out even when the nose is occluded. Toys which motivate the child to carry out many practice trials are useful at this level. As the child becomes used to using the new sound it is easy to involve counting games or many board or lotto games which use the practice sound. Favourite toys include those such as Pop-Up Pirate™, Manic Martians™ and Shark Attack™ (or their many variants). Each turn is taken quickly to allow for repeated drills of the oral sound. 5. Prepare two symbols to indicate ‘mouth sound’ and ‘nose sound’ and introduce listening and discrimination work, using them to build the child’s awareness of the sounds. This will require the therapist, or other adult working with the child, to be able to produce a version of the child’s sound for contrast. 6. When the child is consistently producing the novel sound in isolation, combine it with vowels and then introduce CVC constructions as would be normal in articulation therapy. At this stage the sound can be inserted into nonsense and real words. Children will usually accept the use of the ‘new sound’ without difficulty. The therapist/adult provides reinforcement of the oral sound, and builds awareness of when there has been reversion to the old ‘nose’ sound, i.e., “Was that your old sound, can you fix it?” Commercial card games can be adapted to use the vocabulary being targeted. If working in schools, new words introduced for the current topic are learned with the ‘novel’ sound in place from the start, e.g., an ‘Africa’ topic allows

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‘Masai, savannah, zebra, grassland’, etc., to be added to practice vocabulary for a child. Generalisation into normal speaking situations will require the usual range of creative and individual strategies. At this stage, or sometimes earlier, it is usually possible to shape the sound to something nearer the target, e.g., by encouraging the child to move his/her tongue back, or by reducing the /t/ element.

Older children For some children, especially those who have started reading, explicitly talking about the letter ‘s’ may be necessary. • Introducing the idea of an ‘old’ (nasal) /s/ and a ‘new’ (oral) /s/ can be effective • Initially, as with younger children, parents can be encouraged not to practise the new sound at home, to allow time for it to establish in the therapy setting. In the initial stages of therapy the sound is vulnerable and children easily revert back to their stored motor programme, i.e., ANF substitution • Blowing and oral airstream activities are useful if the child is not stimulable for any novel oral sounds (most often seen with children who have had a lot of unsuccessful therapy) • Try visual and tactile feedback, such as the use of a mirror misting with nasal airflow or the feel of the airstream across the tongue • /θ/ is often the easiest to elicit with older children as it is less likely to feature in their consonant inventory, although confusion with /f/ may need to be addressed.

• Case study: Harry

Harry, aged 3 years and 4 months, presented at clinic with ANF replacing /s, z, ʃ/ sounds. The affricates /ʧ, ʤ/ were stopped to [t, d] respectively. Harry had fluctuating hearing loss during the previous eight months with grommet insertion two months earlier. Hearing levels were within normal limits at his 6-week post-grommet audiology assessment. Harry was very aware that he could not do the ‘snake’ sound. He was anxious and reluctant to try the sound during the assessment session.

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Diagnostic therapy Harry attended a block of once weekly therapy for approximately six weeks. During the first session, games from the magic box (see Chapter 8) were used to encourage gentle oral airstream. Once Harry was more confident, sound pictures were introduced for sounds that he could say, such as /p/. Towards the end of the session the [ts͡] sound was introduced linked to the ‘train slowing down’ picture. Parents were asked to play some of the simple oral airstream activities at home with Harry, but not to try the new [ts͡] sound. In subsequent sessions the [ts͡] sound was practised and established at a single sound level both in therapy sessions and at home, before linking with a vowel within nonsense words. (See ‘road game’ example on the Resource CD.) Verbal feedback from the clinician alluded to the oral/ nasal contrast but it was not taught specifically. For example: “I think that sound disappeared into your nose!” “See if you can make it come out of your mouth, so that you can feel the air.” Deferred responsibility (discussed in Chapter 8) was used to make sure that Harry didn’t give up if he used an ANF instead of his new sound. The concept of the ‘tired tongue’ was used. Harry was encouraged to ‘wake up his tongue’ and ‘see if he could use a lovely train slowing down sound’. Once Harry was using the sound in words such as boots, cats, lets, his parents were encouraged to remind him to use his new sound in his general talking.

Favourite motivating toy Crocodile Dentist (MB Games) was used as a reward for saying the sounds.

Outcome Harry attended for a further two sessions. The new sound was lengthened to isolate the /s/ sound. This was then introduced in a word initial position quite easily. He had a further review three months later and was discharged.  Resource CD: Resources for ANF therapy can be found on the Resource CD.

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Summary Active nasal fricatives can cause speech to sound excessively nasal and impact on intelligibility but are not, by themselves, indicators of poor palate function. In some children the pattern is entrenched and may not correct quickly so it is important that families and schools are aware of this. For a child with VPD who has developed the sound /s/ as a compensatory pattern speech therapy will still be required, even if surgery or prosthetics have resolved the VPD and provided the child with the physical ability to produce target oral sounds. 

References Golding-Kushner, K. (2001). Therapy Techniques for Cleft Palate Speech & Related Disorders. San Diego, CA: Singular. Harding, A. & Grunwell, P. (1998). Active versus passive cleft-type speech characteristics. International Journal of Language & Communication Disorders, 33(3), 329–352. Hattee, C., Farrow, K., Harland, K., Sommerlad, B. & Walsh, M. (2001). Are we ready to predict speech development from babble in cleft lip and palate children? International Journal of Language and Communication Disorders, 36, S1, 115–120. Henningson, G. & Isberg, A. (1988). Influence of tonsils on velopharyngeal movements in children with craniofacial anomalies and hypernasality. American Journal of Orthodontics and Dentofacial Orthopedics, 94, 253–261. Peterson-Falzone, S.J., Hardin-Jones M.A. & Karnell M.P. (2010). Cleft Palate Speech, 4th Edn. St. Louis: Elsevier Mosby. Shprintzen, R., Sher, A. & Croft, C. (1987). Hypernasal speech caused by tonsillar hypertrophy. International Journal of Pediatric Otolaryngology, 14, 45–56. Watson, A., Sell, D. & Grunwell, P. (Eds) (2001). Management of Cleft Lip and Palate. London: Whurr Publishers.

10 Section 3 Electropalatography Lucy McAndrew (Therapy with an electropalatography plate by Helen Piggott)

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Aim of this section • To describe electropalatography (EPG) and how it works. • To explore what EPG tells us about ‘normal’ speech and cleft palate speech. • To discuss whether EPG is useful in assessment and intervention for children and adults with cleft speech characteristics. • To consider what EPG can offer over and above traditional therapy approaches. • To discuss the limitations of and future directions for EPG.

What is EPG? Electropalatography (EPG) is an instrumental computer-based technique which displays real-time visual feedback of tongue to hard palate contact during speech (Hardcastle & Gibbon, 1997; Hardcastle et al., 1991). A custom-made artificial palate is worn by the child or adult. This senses tongue contact in speech through a series of embedded electrodes and corresponding patterns are displayed on a screen. The technique can be used to assess articulatory patterns (spatial and temporal characteristics) and the visual biofeedback can also be used in therapy to help change abnormal patterns. The body of evidence to support EPG’s effectiveness as a diagnostic and therapeutic tool in the treatment of cleft speech difficulties is increasing and many single and multiple case studies report on the benefits of EPG. However, there are limitations within the current literature and a recent Cochrane review concluded that the evidence supporting the efficacy of EPG is not strong (Lee et al., 2009). There are also some drawbacks with its present use.

What does EPG tell us about ‘normal’ speech? EPG is a useful tool because it contributes to our understanding and assessment of articulatory behaviour. It is a safe and objective means of measuring an otherwise inaccessible articulator, the tongue. A number of studies have reported on normative data for tongue-palate contacts. For example, Gibbon et al. (2007a) studied contacts for /t/ and /d/ in comparison to /n/ in normal speakers and showed that all alveolar stops had similar EPG data (i.e., 99% of alveolar stops had anterior constriction with no contact in the posterior

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middle region of the palate). Gibbon et al. (2007b) also looked at tongue-palate contacts for bilabials in normal speakers. They established that bilabial-lingual double articulations that have previously been identified as compensatory articulations (e.g., Trost, 1981), are not a normal feature in English speakers. Tongue-palate contact in vowels has also been investigated in normal speakers by Gibbon et al. (2010). This study concluded that the complete tongue-palate contact which had previously been reported in high vowel production in cleft palate speech (Gibbon et al., 2005) is also not a typical feature of normal English speakers. The findings from the above studies give useful normative adult data. Such data can be used to compare with data from children or adults with cleft speech difficulties. It is important however, to consider that a child (or an adult) with a cleft palate may have different articulatory patterns based upon their age and due to abnormalities relating to their palate shape and dentition. Despite this, such normative data is helpful when assessing the normality or

Figure 10.3.1 An EPG plate Reproduced by kind permission of Articulate Instruments Ltd www.articulateinstruments.com

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abnormality of patterns produced by children or adults with cleft palate. It is also useful to have the visual feedback of ‘normal’ EPG patterns to guide remediation of articulation errors in therapy.

Normative data from speakers with a cleft palate EPG data also provides significant information regarding typical cleft speech articulatory patterns. For example, in one of the largest EPG studies, Yamashita et al. (1992) investigated 53 Japanese speakers with cleft palate and attempted to categorise perceptually judged speech errors on the basis of EPG data. Findings indicated that different tongue-palate contacts resulted in the same perceived type of speech error and that distinct error categories could be defined (palatalised, lateral and nasopharyngeal misarticulations). Overall, broader and more posterior tongue-palate contact in cleft speakers was observed.

What does EPG tell us about cleft palate speech? The diagnostic value of EPG was investigated by Gibbon (2004). Twenty-three articles reporting EPG data over 30 years were reviewed and eight abnormal articulatory patterns in speakers with cleft palate were summarised. These patterns included increased palatal contact, retraction to palatal or velar placement and increased variability. Identification of variability using EPG is particularly useful because, although it is possible to identify this from auditory impression, EPG allows quantification of variability using a variability index (Farnetani & Provaglio, 1991). Thus, the degree of variability over a succession of word repetitions can be captured precisely. This can then be used to monitor progress through therapy. The evidence of such abnormal patterns reported from this extensive literature review of EPG data clearly enhances our knowledge and understanding of the articulatory behaviour in children with cleft speech difficulties.

Hidden articulation? EPG measures articulatory behaviour which is often visually and perceptually inaccessible. It can confirm our perhaps hesitant perceptual judgements. For example, EPG was used by Howard and Pickstone (1995) to confirm their perception that a 6-year old girl with cleft palate was indeed producing alveolar and velar targets with glottal articulation only and not as double articulations. EPG is not only able to confirm or negate perceptual judgements in an objective

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and quantifiable way, but it can also add to the clinical picture by providing more information than we can glean from auditory impression alone. A single case study by Gibbon and Crampin (2001) investigated place of articulation of /t/and /k/ in an adult with a repaired cleft palate. Perceptual analysis suggested both these targets were produced as homophonous middorsum palatal stops. The EPG data, however, revealed that a more posterior placement was used for /k/ than for /t/. As the listener’s analysis alone did not indicate this phonetic difference, this is termed a ‘covert contrast’ (Hewlett, 1988). Identification of covert contrasts is considered important as they indicate the speaker’s awareness of the need to differentiate between sounds. Thus, they signal the difference between a phonetic and a phonological error. EPG can be used as a diagnostic tool to reveal this distinction and such a finding has implications for intervention also, as a phonetic difficulty will respond to an articulatory-based therapy approach. In contrast, a phonological error will need a more linguistic conceptual approach (Sell et al., 1994; Bernthal & Bankson, 1988). The production of alveolar-velar double articulations is an example of a cleft speech error that is frequently misheard as accurate. For example, Hardcastle et al. (1989) reported variability whereby listeners transcribed some /t/ and /d/ productions as accurate and some as [t͡k] or [d͡g]. A study by Gibbon et al. (2004) used EPG data to show that alveolar-velar double articulations were present in 10 out of 15 speakers with a cleft palate despite the fact that no double articulations were identified by therapists from auditory impression alone. Furthermore, the EPG data enabled identification of covert contrasts for one speaker (i.e., a more fronted lateral contact for /t/ and /d/ than for /k/ and /g/). It also highlighted that some of the children achieved alveolar placement alone at least some of the time. Such EPG data are significant not least to add to our current knowledge of tongue placements for these sounds in cleft speech but also for assessment and subsequent tailored intervention targets. For example, in the above study, the infrequent but accurate alveolar placements identified with EPG could then be used in therapy to maximise a child’s ability and to reduce variability using the visual feedback of EPG.

EPG in intervention for children and adults with cleft speech characteristics EPG therapy focuses on changing abnormal articulatory patterns using the visual feedback to guide change (see pp.252–253 for practical application and therapy process). It is particularly useful for children (or adults) with cleft

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palate as many of their speech errors involve tongue-palate contact. Indeed, Lawrence and Philips (1975) reported that around 35% of children with a cleft have speech difficulties affecting alveolar contacts. Encouraging outcomes of EPG therapy have been reported. For example, one survey described therapy delivered in Scotland to 60 children, 27 of whom had a cleft palate (Gibbon & Paterson, 2006). All children had received EPG therapy between 1993 and 2003. Findings indicated that the majority of children (87%) who completed therapy achieved at least some success. Notably, the cleft palate group achieved better overall success rates than those with functional disorders. In addition to positive treatment outcomes, faster progress in therapy using EPG has also been reported (Gibbon et al., 2001; Michi et al., 1993). The benefits of EPG as a therapeutic tool for children with cleft palate is well documented (e.g., Dent et al., 1995; Michi et al., 1993). However, many recent studies report on single (or multiple) case studies (e.g., Lohmander et al., 2010), the findings of which, whilst descriptive in nature, cannot be generalised to other cases.

Longstanding speech difficulties As children become older, articulation difficulties relating to cleft palate are increasingly unlikely to spontaneously resolve or respond to traditional therapy approaches (Nordhoff et al., 1990). EPG has been demonstrated to be particularly useful in treating this group of older children with longstanding intractable speech errors. Whitehill et al. (1996) discussed the use of EPG in the treatment of an 18-year-old Cantonese girl with late cleft palate repair. Results of 13 traditional therapy sessions were reported to be disappointing. However, 23 sessions of EPG therapy resulted in ‘rapid and dramatic improvement’ (Whitehill et al., 1996, p.164) and generalisation to non-target words was also noted. Gibbon and Hardcastle (1989) similarly discussed the success of EPG in the assessment and remediation of a 13-year-old boy with cleft palate where traditional therapy had previously failed. They reported the boy to be at an ‘ideal age to benefit from EPG therapy’ (Gibbon & Hardcastle, 1989, p.108). Thus, a number of single case studies support the view that EPG is particularly useful in treating older children with cleft speech difficulties.

Are there any additional outcomes of EPG therapy? Benefits of EPG therapy other than articulatory change have been noted.

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For example, nasal emission (as a learnt pattern) improved following EPG therapy in the single case study reported by Whitehill et al. (1996). Also, a significant finding of the survey noted above (Gibbon & Paterson, 2006) was that almost all therapists (93%) considered that the children treated with EPG showed increased awareness of their speech difficulties. The biofeedback that is unique to EPG allows the child to pattern match and to visualise incorrect compared with correct articulations, thereby increasing their awareness of their difficulties.

Generalisation of speech sounds Generalisation of newly-acquired speech sounds in a controlled setting using EPG, as with any traditional therapeutic approach, can prove challenging. The survey by Gibbon and Paterson (2006) reported that 88% of the group surveyed had at least some difficulty with generalisation of newly-acquired patterns. Their findings support the view that EPG is most useful in the establishment phase of therapy as discussed by Smit (2004) and that consequently there is a need to address generalisation and maintenance of new sounds following EPG therapy. This view is also supported by Lohmander et al. (2010) who discussed difficulties with the transfer from function (articulation) to activity (intelligibility) in reference to a single case study. Future research into the measurement of generalisation and maintenance following EPG therapy is recommended (Smit, 2004).

How might EPG therapy be successful when traditional methods are not? EPG can be used to change articulatory behaviour when traditional therapeutic approaches have proved unsuccessful and this may be due to a number of factors. The immediate visual feedback enables speech patterns to be tangible for children and adults. It provides objective, measurable feedback enabling the child or adult to make and note small changes in their articulatory behaviour. In particular, children can work more actively and independently in therapy thereby increasing their motivation, and they often enjoy and respond to the novelty of working with a computer-based system. However, not all studies report successful outcomes following therapy using EPG. McAuliffe and Cornwell (2008) describe the use of EPG in the remediation of lateral /s/ in a single case study. Although functional improvements in /s/ were perceived,

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they found that only minimal changes in EPG patterns were evident following intervention. They consequently recommended the combined use of EPG therapy to initiate an accurate articulatory pattern followed by an intensive traditional approach. This study supports the view that EPG should not be used instead of traditional approaches but rather as an additional tool when necessary (Gibbon & Hardcastle, 1987).

Accessibility of EPG Despite the many benefits that EPG has to offer, a significant barrier to its use is cost and accessibility. Palate manufacture is expensive and time-consuming. Data analysis also requires time and expertise. To help overcome inaccessibility, CLEFTNET Scotland was developed in 1996 (Gibbon et al., 1998). This network linked cleft palate centres in Scotland electronically to EPG experts at Queen Margaret College, Edinburgh. EPG data could then be transferred to experts for analysis, and results together with therapy advice could then be returned. This provision now extends to all UK cleft centres so guidance on EPG therapy is now widely available to cleft specialists (CLEFTNET UK; Lee et al., 2007). The provision of EPG portable training units (PTUs) has additionally enabled increased access (Fujiwara, 2007; Gibbon et al., 1999). These relatively lightweight and inexpensive units permit home practice, thereby increasing the child or adult’s frequency and control over their own therapy and potentially reducing long travel commitments to attend therapy sessions.

Limitations of EPG By no means is EPG therapy useful for all children and adults with cleft speech difficulties. It has successfully been used with persisting and intractable speech difficulties but there are other factors that can affect its usefulness. Acclimatisation and tolerance of the artificial palate is not always straightforward. Changes in oral cavity growth and dentition can also affect accurate palate fitting. In an investigation of EPG treatment in school-aged children and young adults, two out of 23 subjects did not complete therapy due to palate intolerance and a lost upper tooth onto which the appliance was fixed (Dent et al., 1995). An ability to comprehend the visual display relative to the auditory stimuli is also a prerequisite for effective EPG treatment. The recent Cochrane review highlights that ‘a certain level of cognitive abilities of the individuals is required in order for them to benefit from EPG therapy’ (Lee et al., 2009, p.4).

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Candidate selection The age, maturity, awareness, level of concern and motivation of the child or adult, together with the type and severity of speech problem, are all factors which will impact on the success of EPG in remediation. Similarly, parental/ carer support and the therapist’s knowledge, experience, expectations and ability to motivate will all impact on outcomes. Thus, as many authors recommend, care should be taken in assessing subject selection for EPG therapy (Lee et al., 2007; Dent et al., 1995; Morgan Barry, 1989).

Technical limitations Despite the unique ability of EPG as a diagnostic and remediation tool, it does present some limitations. EPG gives information regarding placements on the hard palate. Other articulations either more anteriorly (e.g., dental or labial articulations) or more posteriorly (e.g., uvular, pharyngeal or glottal articulations), however, are not recorded. Even velar articulations are variably recorded depending on the individual speaker and surrounding vowels. Similarly, although hard palate placements are registered, EPG does not inform regarding manner of articulation (e.g., plosive, fricative or nasal). EPG can tell us what is happening regarding tongue-palate contact, but it does not tell us why it is happening. Such limitations illustrate why EPG is useful as a supplementary tool alongside perceptual assessment and case history details. There are a number of other technical limitations. For example, with the Reading palate (Hardcastle et al., 1991) which is now widely used in the UK, electrodes placed closely together can be switched on and off by the presence of saliva (Wrench, 2007). The thickness of the palate can have an impact on articulation (McLeod & Searl, 2006) and no design has yet been developed which does not need leads to extend from the mouth. Further issues include: the sampling rate of 100 Hz, which is often inadequate for research; the software and hardware which are not compatible with some new computers; and the PC-based system which is not easily transportable. All of the above illustrate that there are economic, clinical and technical issues with the current use of EPG.

Future directions for EPG Although there is accumulating evidence for normative EPG data of ‘typical’ English speakers, there is a lack of data for ‘normal’ children and adults with clefts (i.e., those with no speech errors). Such data would enable more useful

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comparisons for children or adults with cleft speech errors in assessment and therapy. A particular area that has so far received little attention is how EPG can provide significant information regarding abnormal timing of articulations. One study identified a covert contrast in a 6-year-old girl’s production of /t/ and /k/ versus /d/ and /g/ in that the EPG data highlighted a longer closure phase before the aspirated release of the /t/ and /k/ in comparison to /d/ and /g/ (Howard, 1993). The importance of timing of articulations is an area requiring further investigation and larger-scale studies. In many EPG studies, sound segments are identified and investigated in isolation (e.g., Gibbon & Crampin, 2002). However, there is a paucity of research investigating connected speech processes in children with cleft palate using EPG. Howard (2004) investigated five older children with intractable speech disorders (two of whom had cleft palate) and found that all children produced atypical (in addition to typical) articulatory behaviours at syllable boundaries. The author stressed the importance of using instrumental analysis such as EPG to identify normative connected speech data in order to draw comparisons with data derived from children with speech difficulties. In terms of EPG instrumentation, Scobbie et al. (2004) presented a new generation of hardware and software compatible with the Reading system which provides more accessible and powerful tools in the analysis of articulatory and acoustic data. Improvements include ability to load EPG hardware onto a laptop and loading and adjusting target patterns to suit individual needs. The integration of ultrasound (Stone, 1997) and high quality audio-feedback and video with EPG is a future innovation hoped to reveal for example, the posterior articulatory behaviours which are not seen with the current EPG system. Improved palate design is also imminent. Wrench (2007) introduced a new palate, the Articulate palate, which is reported to have many new benefits. These include significantly reduced cost, improved circuit design and electrode arrangement and a thinner but tighter palate fitting resulting in improved acclimatisation and comfort. Finally, the recent Cochrane review of EPG for articulation disorders associated with cleft palate (Lee et al., 2009) systematically surveyed the literature and found the evidence base for the effectiveness of using EPG in treating children and adults with cleft speech errors not to be strong. Despite numerous studies based on low numbers or single cases, they reported a lack of high-quality large-scale randomised control trials (RCTs). The emergence of CLEFTNET UK however, is an ideal infrastructure for carrying out such RCTs in the future.

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Summary and conclusion In summary, EPG can be a very useful additional tool in the assessment of and intervention for children and adults with cleft speech difficulties. The following advantages have been discussed in this section: • EPG enhances the comprehensiveness of assessment and can provide insights into articulatory behaviour which are sometimes hidden in perceptual analysis alone • Data interpretation can result in more accurate diagnosis which can then lead to tailored and consequently more effective treatment • EPG provides a unique biofeedback system which allows small observable changes to be made • EPG provides a novel, visual and appealing approach for children and adults when traditional therapy may have been unsuccessful • EPG has been demonstrated to be particularly useful with older children with long-standing intractable cleft speech difficulties. Many factors need to be considered in subject selection, and generalisation and maintenance of newly-acquired skills requires further attention. Despite its many benefits, there are a number of limitations with the current use of EPG, cost and inaccessibility being significant drawbacks. However, recent developments in system and palate design are promising. Finally, although single case studies report individual benefits, such findings cannot be generalised and therefore larger-scale RCTs are essential in demonstrating the usefulness of EPG in the management of children and adults with cleft speech difficulties.

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Therapy with an electropalatography (EPG) plate Helen Piggott EPG is often thought of as a last resort to remediate articulation problems because of the cost and equipment implications. However, its use can be very effective, producing good results in a relatively short space of time. The use of EPG within the context of articulation assessment and therapy and the supporting evidence base has been reviewed in this chapter. We will now consider how to introduce and use EPG in the clinical setting.

Steps to successful EPG therapy It is preferable for the clinician to have their own EPG plate but not essential. It is unlikely that teaching support staff or parents will have a plate, and yet will be carrying out the EPG practice with the child. Successful outcomes can be achieved with the child’s EPG plate only. 1. Ensure that the plate is fitting comfortably. The child and adults working with the child should be comfortable with putting the plate in and taking it out; understand how the EPG works and how to connect everything together; and be aware of the hygiene and safety issues surrounding its use. These will be explained in detail by the cleft centre SLT who is overseeing the therapy. 2. Ask the child to experiment with moving their tongue around in their mouth making contact with the plate, to see which lights on the display are illuminated. Try all lights lit, a few, none, one side, then the other side, etc. For example: • no contact – no light lit • full contact – all lights lit • tip of tongue – just a few lights at front lit. Encourage the child to describe what they are doing with their tongue to create that pattern of lights/contacts. This is a good time to talk about how much of the tongue is coming into contact with the plate and to talk (in simple terms) about the ‘lateral bracing’ (the ‘bridge’

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or ‘arch’ shape) which occurs in the production of many consonants (/t,d,s,z,ʃ/ etc.). 3. Often the aim of using EPG is to achieve improved articulation for alveolar sounds. The first step is to name the sequence of lights that will appear when alveolar placement has been achieved. This is easier if the clinician has a plate as they can model the /t/ sound silently and show the pattern.

The pattern for alveolar plosives looks like a ‘bridge’ or ‘arch’ and changes to an ‘open bridge’ for /s/ and /z/, i.e., with a central groove to allow release of the fricative sound. However, the child could come up with their own terms if they so prefer. Avoiding naming or writing the target sounds prevents the child from reverting back to their stored motor programme. It can also be helpful with children with an ingrained pattern such as backing, to introduce other alveolar consonants first, e.g., /n/ or /l/, or introduce them as a group. This helps to establish the ‘bridge’ pattern, and to help break the ‘I can’t do that sound’.

4. Ask the child to practise the ‘bridge’ pattern silently, ensuring that they are making an accurate pattern of contacts using the visual display. 5. Then introduce single sound production, again maintaining the ‘bridge’ pattern. Practising with eyes closed can help the child really think about where they are putting their tongue. They then open their eyes and see if the picture appears correct by checking the visual display. 6. Continue, as with any other articulation work, gradually building the sound into CV and VC words and beyond. Consider carefully any other consonants that are included in these early words and the vowels that are used, as these could help or hinder accurate production. For example, using the high vowel /i/ with /t/ can sometimes be helpful. 7. Once the child is maintaining accurate production in simple words, it is a good idea for them to have a short period of practice, producing the new sound without the EPG plate in. This is best carried out immediately after the session whilst placement is fresh in their mind. Time without the plate is gradually increased, providing the child continues with accurate production.

8. Clinicians should be clear about when and how much practice the child should do with the new sound outside of their EPG sessions. As with all newly-acquired sounds they are very vulnerable to

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old habits creeping back in. Parents and schools are advised to aim for ‘little and often’ practice sessions to allow for the frequent repetitions needed for learning. 9. Generalisation work is carried out in the same way as with other sounds. EPG is mostly carried out to facilitate accurate production of the single sound, but can then be used to establish the new correct sound into words. The approach tends to be less helpful beyond short phrases, as the visual feedback becomes confusing and therefore less effective.

References Bernthal, J.E. & Bankson, N.W. (1988). Articulation and Phonological Disorders. Englewood Cliffs, NJ: Prentice Hall. Dent, H., Gibbon, F. & Hardcastle, B. (1995). The application of electropalatography (EPG) to the remediation of speech disorders in school-aged children and young adults. European Journal of Disorders of Communication, 30, 64–277. Farnetani, E. & Provaglio, A. (1991) Assessing variability of lingual consonants in Italian. Quaderni del Cantro di Studio per le Ricerche di Fonetica del C.N.R, X, 117–145. Fujiwara, Y. (2007). Electropalatography home training using a portable training unit for Japanese children with cleft palate. International Journal of Speech-Language Pathology, 9, 65–72. Gibbon, F. & Hardcastle, W. (1987). Articulatory description and treatment of ‘lateral s’ using electropalatography: A case study. British Journal of Disorders of Communication, 22, 203–217. Gibbon, F. & Hardcastle, W. (1989). Deviant articulation in a cleft palate child following late repair of the hard palate: A description and remediation procedure using electropalatography. Clinical Linguistics and Phonetics, 3, 93–110. Gibbon, F., Crampin, L., Hardcastle, B., Nairn, M., Razzell, R., Harvey, L. & Reynolds, B. (1998). CLEFTNET Scotland: A network for the treatment of cleft palate speech using EPG. International Journal of Language and Communication Disorders, 33 (supplement), 44–49. Gibbon, F., Stewart, F., Hardcastle, W. & Crampin, L. (1999). Widening access to electropalatography for children with persistent sound system disorders. American Journal of Speech and Language Pathology, 8, 319–334. Gibbon, F.E. & Crampin, L. (2001). An electropalatographic investigation of middorsum palatal stops in an adult with repaired cleft palate Cleft Palate-Craniofacial Journal, 38, 96–105. Gibbon, F., Hardcastle, W.J., Crampin, L., Reynolds, B., Razzell, R. & Wilson, J. (2001). Visual feedback therapy using electropalatography (EPG) for articulation disorders associated with cleft palate. Asia Pacific Journal of Speech, Language and Hearing, 6, 53–58.

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Gibbon, F.E. & Crampin, L. (2002). Labial-lingual double articulations in speakers with cleft palate. Cleft Palate-Craniofacial Journal, 39, 40–49. Gibbon, F. (2004). Abnormal patterns of tongue-palate contact in the speech of individuals with cleft palate Clinical Linguistics & Phonetics, 18, (4–5), 285–311. Gibbon, F., Ellis, L. & Crampin, L. (2004). Articulatory placement for /t/, /d/, /k/ and /g/ targets in school age children with speech disorders associated with cleft palate. Clinical Linguistics and Phonetics, 18, 391–404. Gibbon, F., Smeaton-Ewins, P. & Crampin, L. (2005). Tongue-palate contact during selected vowels in children with cleft palate. Folia Phoniatrics Logopedics, 57, 181–192. Gibbon, F. & Paterson, L. (2006). A survey of speech and language therapists’ views on electropalatography therapy outcomes in Scotland. Child Language Teaching and Therapy, 22, 275–292. Gibbon, F., Yuen, I., Lee, A. & Adams, L. (2007a). Normal adult speakers’ tongue palate contact patterns for alveolar oral and nasal stops. International Journal of Speech-Language Pathology, 9, 82–89. Gibbon, F., Lee, A. & Yuen, I. (2007b). Tongue-palate contact during bilabials in normal speech. Cleft Palate-Craniofacial Journal, 44, 87–91. Gibbon, F., Lee, A. & Yuen, I. (2010). Tongue-palate contact during selected vowels in normal speech. Cleft Palate-Craniofacial Journal, 45, 405–412. Hardcastle, W., Morgan, Barry R. & Nunn, M. (1989). Instrumental articulatory phonetics in assessment and remediation: Case studies with the electropalatograph. In J. Stengelhofen (Ed.) Cleft Palate: The Nature and Remediation of Communicative Problems (pp.136–164). Edinburgh, Scotland: Churchill Livingstone. Hardcastle, W.J., Gibbon, F.E. & Jones, W. (1991).Visual display of tongue-palate contact: Electropalatography in the assessment and remediation of speech disorders. British Journal of Disorders of Communication, 26, 41–74. Hardcastle, W.J. & Gibbon, F. (1997). Electropalatography and its clinical applications. In M.J. Ball & C. Code (Eds), Instrumental Clinical Phonetics (pp.149–193) London: Whurr Publishers. Hewlett, N. (1988). Acoustic properties of /k/ and /t/ in normal and phonologically disordered speech. Clinical Linguistics and Phonetics, 2, 29–45. Howard, S. (1993). Articulatory constraints on a phonological system: A case study of cleft palate speech. Clinical Linguistics and Phonetics, 7, 299–317. Howard, S. & Pickstone, C. (1995). Cleft palate: perceptual and instrumental analysis of a phonological system. In M. Perkins and S. Howard (Eds), Case Studies in Clinical Linguistics (pp.65–90). London: Whurr Publishers. Howard, S. (2004). Connected speech processes in developmental speech impairment: Observations from an electropalatographic perspective. Clinical Linguistics and Phonetics, 18, 405–417. Lawrence, C.W. & Philips, B.J. (1975). A telefluoroscopic study of lingual contacts made by persons with palatal defects. Cleft Palate Journal, 12, 85–94.

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Lee, A.S.Y., Gibbon, F., Crampin, L., Yuen, I. & McLennan, G. (2007). The national CLEFTNET project for individuals with speech disorders associated with cleft palate. Advances in Speech-Language Pathology, 9(1), 57–64. Lee, A.S.Y., Law, J. & Gibbon, F. (2009). Electropalatography for articulation disorders associated with cleft palate. Cochrane Database of Systematic Reviews, Issue 3. Lohmander, A., Henriksson, C. & Havstam, C. (2010). Electropalatography in home training of retracted articulation in a Swedish child with cleft palate: Effect on articulation pattern and speech. International Journal of Speech-Language Pathology, 12(6), 483–496. McAuliffe, M.J. & Cornwell, P.L. (2008). Intervention for lateral /s/ using electropalatography (EPG) biofeedback and an intensive motor learning approach: A case report. International Journal of Language and Communication Disorders, 43, 219–229. McLeod, S. & Searl, J. (2006). Adaptation to an electropalatograph palate: Acoustic, impressionistic, and perceptual data. American Journal of Speech-Language Pathology, 15, 192–206. Michi, K., Yamashita, Y., Imai, S., Suzuki, N. & Yoshida, H. (1993). Role of visual feedback treatment for defective /s/ sounds in patients with cleft palate. Journal of Speech and Hearing Research, 36, 277–285. Morgan, Barry R. (1989). EPG from square one: An overview of electropalatography as an aid to therapy. Clinical Linguistics and Phonetics, 3, 81–91. Noordhoff, M.S., Huan, C.S. & Wu, W.T. (1990). Multidisciplinary management of cleft lip and palate in Taiwan. In J. Bardach & H.L. Morris (Eds), Multidisciplinary Management of Cleft Lip and Palate (pp.18–26). Philadelphia, PA: W.B. Saunders. Scobbie, J.M., Wood, S.E. & Wrench, A.A. (2004). Advances in EPG for treatment and research: An illustrative case study. Clinical Linguistics and Phonetics, 18, 373–389. Sell, D., Harding, A. & Grunwell, P. (1994). A screening assessment of cleft palate speech (Great Ormond Street Speech Assessment). European Journal of Disorders of Communication, 29, 1–15. Smit, A. (2004). Articulation and Phonology: Resource Guide for School-Age Children and Adults. New Jersey: Thomson Delmar Learning. Stone, M. (1997). Laboratory techniques for investigating speech articulation. In W.J. Hardcastle and J. Laver (Eds), The Handbook of Phonetic Sciences (pp.11–32). Oxford: Blackwell. Trost, J.E. (1981). Articulatory additions to the classical description of the speech of persons with cleft palate. Cleft Palate Journal, 18, 193–203. Whitehill, T.L., Stokes, S.F. & Yonnie, M.Y. (1996). Electropalatography treatment in an adult with late repair of cleft palate. Cleft Palate-Craniofacial Journal, 33, 160–168. Wrench, A.A. (2007). Advances in EPG palate design. Advances in Speech-Language Pathology, 9, 3–12. Yamashita, Y., Michim K., Imai, S,. Suzuki, N. & Yoshida, H. (1992). Electropalatographic investigation of abnormal lingual-palatal contact patterns in cleft palate patients. Clinical Linguistics and Phonetics, 6, 201–217.

10 Section 4 Speech Therapy and Other Non-Surgical Treatments for Nasal Speech Ginette Phippen

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Aim of this section This section reviews the literature relating to speech therapy and other non-surgical interventions for nasal speech and discusses whether there is a place for speech therapy in the battery of treatments for this speech disorder. One specific treatment is explored in more detail, Visual Feedback Therapy, including theoretical rationale and procedure, as well as results of preliminary evaluation of this approach.

Introduction The past 50 years have seen significant developments in the frameworks for assessing the complex speech mechanism and identifying the contributory components in speech disorders associated with cleft and Velopharyngeal Dysfunction (VPD) (Kuehn & Moller, 2000). However, there remains a lack of consensus about the role of speech therapy and other non-surgical treatments in treating nasal speech. There is some evidence that speech therapy combined with surgery can be the most effective management approach (Mercer & Pigott, 2001) generally to address compensatory articulation patterns, but the use of speech therapy as a primary treatment for nasal speech is not widely supported. Nonetheless, speech therapy treatment of nasal speech continues to attract interest and debate as there is very limited evidence of efficacy. Historically, a variety of interventions have been offered for nasal speech including palate exercises, stimulation, resistance training, visual biofeedback, articulation therapy and voice therapy. These types of behavioural interventions may or may not be defined as a speech therapy intervention, but all operate on the premise that new motor speech behaviours involving the soft palate can be learned as long as there is sufficient palate tissue to close the mouth off from the nose. In addition, there must be adequate muscle and nerve function for movement of the soft palate. Three phases of opinion can be identified relating to the effectiveness of such speech therapy treatment (Kuehn & Moller, 2000; Tomes, 1997). In phase one (1940s to 1960s) the accepted view was that speech therapy was effective. In phase two (1960s to 1970s) the view was that speech therapy does not work for nasal speech but can improve articulation and speech intelligibility generally. The third phase (1970s to 2000 onwards) is that speech therapy might work for nasal speech but that more data are needed on the efficacy of specific

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treatments. Despite a resurgence of interest in non-surgical treatments for nasal speech in the late 1970s, speech therapy was not widely reintroduced as a primary treatment due to the general belief that this approach was ineffective. Unsurprisingly, this view has persisted over the last 30 years (Shprintzen & Marrinan, 2009; Russell, 2006; RCSLT, 2005; Golding-Kushner, 2001) as there continues to be insufficient supporting evidence.

What is the evidence?

• Research

A recent systematic review carried out by Bessell et al. (2012) examined the evidence for speech therapy in children with cleft palate. The inclusion criteria of the Bessell review was any speech therapy approach, at any age, time and setting. Single case studies and studies with five participants or less were excluded, as were studies that included subjects other than children with cleft palate and those with no reported speech outcome. This resulted in only six papers being identified from 1241 citations as meeting these criteria, highlighting the extremely limited evidence base in this area. Adopting a broader approach than the above systematic review does allow for the inclusion of all relevant studies reporting on speech therapy and other non-surgical treatment approaches for nasal speech, irrespective of sample size or outcome measures. However, the evidence is confined to small studies with poorly developed interventions, often lacking in theoretical basis. In addition, outcome measures are not tested for reliability and validity, or even fail to include a perceptual measure of speech. Control of other confounding treatments is inconsistently addressed so that currently it is unclear what is effective, and for whom. It is clear that there is insufficient evidence to support an immediate change in practice at this point. To date, no study has provided unequivocal support for the use of a particular speech therapy or other non-surgical treatment for nasal speech and the majority of approaches have not been studied rigorously. Consequently, although there is consensus that speech therapy and other nonsurgical approaches alone cannot address a structural palate defect (Ruscello, 2006; Dworkin et al., 2004; Kummer, 2001), uncertainty remains about the possible benefits of some interventions for individuals with nasal speech.

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An intervention for nasal speech and how it might work The ultimate goal of any intervention for nasal speech must be a functional improvement in speech, with no adverse effects (Boseley et al., 2004; Johns et al., 2003). Where speech therapy and other non-surgical interventions are described it is therefore vital to consider the underlying mechanisms which may be operating, as well as the aim of the intervention, which may be: • To change the velopharyngeal muscles (strength/mass/endurance) • To change control of the velopharyngeal muscles (coordination/ consistency) • To change other speech factors to reduce nasal speech quality (respiratory/laryngeal/articulatory). Cole (1979) proposed the terms direct and indirect to differentiate between approaches designed either to stimulate or to influence velopharyngeal function. Cole’s definitions are useful in that they allow for consideration of the mechanisms underlying the intervention. This refers to the mechanism by which functional improvement in speech might occur. Direct interventions are based on the premise that muscle training or stimulation techniques can increase the activity of the muscles involved in velopharyngeal closure. In direct treatment approaches, there is an assumption that changes will occur in palate muscle function, for example, increased strength, tone or elasticity. Indirect intervention refers to an approach designed to influence velopharyngeal function but one that does not focus specifically on muscle activity. With indirect approaches, the change is a behavioural one with increased voluntary control over palate movement and associated nasal speech. Speech therapy, including the use of feedback, generally falls into this category.

Direct interventions Exercises are a direct intervention using stimuli such as blowing, sucking, swallowing and gagging and have been used to treat nasal speech. They are classed as non-speech oral motor exercises (NSOMEs) (Bowen, 2005), in contrast to speech therapy approaches that use speech stimuli to influence parameters such as rate, clarity and fluency. The use of non-speech exercises is based on physical therapy principles of exercise, involving practice and repetition and

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includes active muscle exercise and passive muscle exercise. Active exercise involves strength training and stretching whilst passive exercise is the movement of a muscle or muscle group assisted by another person or a machine. The focus is generally on active exercise in relation to palate function. Several authors hypothesise regarding the effect of such exercises on the palate, for example Witt et al. (1995) suggest that a muscle tends to assume the length in which it is habitually maintained so that actively exercising the palate muscle could result in increased stretch or extension during speech. The use of non-speech tasks to influence palate function or improve speech has been largely discredited (Bunton, 2008; Bowen, 2005; Kuehn & Moller, 2000). Ruscello (2008) comprehensively reviews this area and concludes that muscle treatments are not an effective approach for most clients with VPD. The rationale for this is that the underlying mechanisms controlling speech and non-speech palate function differ, with different patterns of palate closure evident in each (Kummer, 2008). Bunton (2008) in particular effectively challenges the assumption that movement characteristics and task demands are similar for speech and non-speech oral behaviours. The author goes on to assert that this is supported by motor learning theory of task specificity, as well as differences in neural organisation demonstrated by MRI studies in healthy individuals. Palate stimulation is a direct intervention based on physiological principles and aims to increase blood supply to the palate in order to reduce fatigue and increase muscle bulk, tone and stretch (Kuehn et al., 2002). Approaches have tended to use either electrical or tactile stimulation, or a combination of both. Peterson (1974) hypothesised that electrical stimulation might result in increased firing of nerve fibres, therefore constituting a type of ‘active’ exercise for the palate in order to stimulate movement. The studies in this area are over 40 years old and since then the use of palate stimulation has been largely abandoned. The evidence for stimulation is therefore very weak and further compromised by methodological weaknesses. Resistance training is a direct intervention that describes the requirement of muscles to move against an opposing force, usually provided by equipment of some kind (Fleck & Kraemer, 2004). The aim of this approach is to create resistance between the device and muscular forces in the palate and so improve velopharyngeal function. Approaches have included bespoke inflatable devices, nose-holding to introduce positive pressure into the oral cavity and the use of pressurised air with Continuous Positive Airway Pressure (CPAP) equipment.

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CPAP appears to offer potential benefit for some individuals with relatively recent studies demonstrating positive results following treatment (Cahill et al., 2004; Kuehn et al., 2002). Participants in these studies included adults with nasal speech resulting from traumatic brain injury as well as children and adults with cleft palate. This suggests that more research is needed to establish clearer candidate suitability. Prosthetic training is a direct intervention that involves the speaker wearing a bespoke dental appliance in the mouth that fixes on to the upper teeth and extends back to touch the soft palate (Ruscello, 1982). A reduction programme is employed to stimulate the muscles and nerves involved in palate closure, with the premise that duration and size of the prostheses are gradually reduced as compensation of velopharyngeal musculature occurs. The limited evidence available suggests that the use of prosthetics to actively stimulate palate function and reduce nasal speech is not effective. However, as with the other types of direct interventions described it is difficult to draw firm conclusions due to the methodological weaknesses of previous studies. See Chapter 4 for details of how prosthetic palatal appliances are currently routinely used as a treatment option for VPD/nasal speech but not usually as a direct intervention designed to stimulate the palate.

In summary… The lack of recent studies in most of the above areas reflects a move away from the theoretical perspective of treating the palate muscles as any other muscle in the body, i.e., as amenable to training to improve strength, range, speed or precision of movement. However, it should be noted that such regimes have not been systematically evaluated in large numbers of individuals and with effective control groups. Nevertheless, it is generally accepted in clinical practice that these direct approaches are not appropriate in treating nasal speech (Peterson-Falzone et al., 2006).

Indirect interventions Biofeedback is an indirect intervention which provides information regarding speech performance to the individual via a signal from an external source. This enables the individual to attempt to volitionally modify their performance by using information from his or her own body (Laures & Shisler, 2004; Earles et al., 2003). Biofeedback approaches to treating nasal speech have taken two forms: direct visualisation of the velopharyngeal structures using a rigid or flexible

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nasendoscope and visual biofeedback generated by specialised instrumental equipment. Brunner et al. (2005) give a useful theoretical breakdown of nasendoscopic biofeedback into four areas as follows, highlighting the learning process involved: • Introduction to structures and function • Experimentation – self-monitoring with guidance • Coupling – anchoring motor and auditory perception • Automatisation – overlearning. They propose that the concepts of improved self-monitoring and improved sensory control are central to the process of nasendoscopic biofeedback and that in order for palate closure to be improved there must be evidence that this is achievable. The same principles can be seen to apply to instrumental biofeedback where physiological output, in this case airflow associated with speech, generates a visually displayed signal. This allows the speaker to monitor their speech and attempt to modify it according to defined parameters or thresholds. Results from studies where visual biofeedback is a component are variable but positive results are reported for some subjects. This suggests that biofeedback may offer potential as a treatment modality for nasal speech. However, there has been only one study, using nasendoscopic biofeedback undertaken in the last 10 years (Brunner et al., 2005). Nasendoscopy is routinely available at regional cleft palate/VPD treatment centres and more speech and language therapists are now independently undertaking this procedure (Sell et al., 2008). This suggests that there is potential for further investigation of this treatment approach. Similarly, there are existing instrumental devices, e.g., nasometry, which can provide visual biofeedback specifically in relation to nasal speech and which are also widely available at the treatment centres. More rigorous investigation of either or both of these biofeedback approaches could be undertaken with the focus on a clearly defined protocol and comprehensive perceptual and instrumental assessment and analysis. Articulation therapy is a generic term for a range of indirect approaches used to treat speech disorders relating to abnormal structure and/or faulty learning of speech sounds (Bowen, 2009). Articulation therapy is essentially based on behavioural principles of shaping a desired response through modelling and reinforcement. In practice, however, as Bowen explains, clinicians draw upon a

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number of theoretical learning frameworks including those relating to speech at the cognitive and linguistic level. The specific effect of articulation therapy in treating nasal speech is unclear, although the consensus is that there is no evidence to suggest articulatory treatment results in improvement in velopharyngeal function (Kuehn & Moller, 2000). It is clear, however, that there is a relationship between articulatory development and VPD (Pamplona, 2000), with certain articulation disorders considered to be compensatory behaviours secondary to palate dysfunction. Voice therapy is an indirect approach that aims to modify speech production, predominantly through behavioural shaping and reinforcement. Treatment programmes vary but can include a focus on increasing vocal awareness, postural adjustment, targeted relaxation, breathing and patterns of voice production based on physiological principles (Cavalli, 2011). Many voice therapy programmes will also pay attention to psychosocial aspects that may be impacting on the physiology of the voice (Ramig & Verdolini, 1998). Cavalli (2011) states that voice therapy may be considered appropriate for individuals who have abnormal voice associated with VPD, e.g., dysphonia characterised by hoarseness, breathiness and strain. It is not widely used for treating individuals with nasal-sounding speech, despite this being a perceived characteristic of voice. This is probably because, as with other non-surgical treatments, there is an assumption that such an approach is ineffective. However, its use has not been extensively investigated with this population and therefore evidence of efficacy is lacking.

In summary… Despite the weak evidence overall the literature does lend some support to the use of visual biofeedback and resistance training approaches, in that observable changes in palate function and reduction of nasal speech characteristics have been reported for some individuals. These findings suggest that further investigation is warranted and that some individuals could have the potential for functional speech improvement using behavioural techniques. Kummer (2002) proposes that speech therapy could be considered as an appropriate treatment: • If the nasal speech characteristics are mild and inconsistent • If the individual can achieve reduction of the characteristic (is stimulable)

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• If the characteristic is caused by faulty use (mislearning) of the velopharyngeal structure or associated with oral motor dysfunction. The above criteria could therefore be defined as the individual demonstrating the necessary potential for velopharyngeal closure. Similarly, several other authors acknowledge the role of speech therapy for selected individuals, usually those with mild and/or inconsistent symptoms or where there is evidence of potential for closure of the palate against the back of the throat (Ruscello, 2008; Ysunza et al., 1997). Jones et al. (2004) also describes a category of patients with ‘borderline’ velopharyngeal competence and again it could be argued that in such cases variation in nasal speech may indicate potential for improvement using speech therapy. The use of behavioural interventions is also supported by research in other areas of speech therapy including articulation and voice disorders, and dysphagia (Marchant et al., 2008; Hagg & Larsson, 2004; Roy & Weinrich, 2003) and this related literature provides a stronger evidence base for the use of such techniques, albeit in different conditions.

Feedback in speech therapy Feedback techniques are described across a wide range of speech therapy client groups and disorders. Feedback may given verbally by the speech therapist in response to the individual’s communication behaviour in relation to specified targets (performance feedback) or visually through the use of instrumentation and computer technology in response to physiological stimuli from the individual (visual biofeedback). Biofeedback tends to be used in more specialist settings, where equipment such as electropalatography is available. Performance feedback, on the other hand, is an intrinsic part of most speech therapy interventions. Types of intervention reported in the literature include electropalatography, ultrasound, electromyography, spectral feedback, electroglottography, Respitrace, electromagnetic articulograph, transnasal flexible laryngoscopy, performance feedback and speech therapy (including feedback). Studies cover a range of disorders, client groups and ages with a focus on speech disorder (voice and articulation) and aetiologies including cerebral palsy, hearing impairment, dysarthria (muscle weakness), cleft palate, Down’s syndrome and vocal misuse. Many studies report positive results following speech therapy intervention using feedback. There are, however. methodological limitations which mean

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that again this can only be taken as indication of a trend rather than evidence of efficacy. As in the literature relating to treatments for nasal speech, many of the studies are single case studies or small case series with no control or comparison, so that any positive results cannot be attributed to the intervention. However, the evidence is more recent and the studies better quality suggesting that this approach does appear worthy of further investigation.

Development of a feedback intervention for nasal speech The lack of strong evidence to support behavioural treatments for nasal speech means that any new intervention needs to be rigorously evaluated before being recommended for routine clinical use. The current author has carried out feasibility testing of such an intervention, known as Visual Feedback Therapy (VFTh). The results of a small study (Phippen, 2013, unpublished) tentatively support the view that some individuals can modify habitual patterns of nasal speech (hypernasal vowels) following therapy using feedback techniques. Further larger-scale research is now needed into the efficacy of VFTh to determine whether this is an intervention worth pursuing. VFTh was developed to include two distinct but interrelated components; the central component is visual biofeedback supported by the addition of performance feedback: • Visual biofeedback is provided in response to the nasal escape of air during speech through the use of visual feedback tools • Performance feedback is direct and targeted verbal feedback given by a speech and language therapist in response to speech behaviour.

Visual biofeedback tools The biofeedback tools used in the author’s study of VFTh were nasometry, See-Scape™, and a nasal mirror, as shown in Figure 10.4.1. These are generally available in the regional cleft palate centre. Nasometry was the main tool used with See-Scape™ and the mirror used to demonstrate gross features of oral and nasal airflow.

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• Terminology

Nasometry is an instrumental tool that gives specific visual feedback of the oral versus nasal balance of speech output via a computer screen. Nasometry, when used as a treatment tool, provides immediate visual feedback about the degree of nasal speech, in picture and graph form, via a computer. There are also colourful and motivating games which again provide immediate visual feedback if speech production reaches a pre-set target, for example below a certain percentage nasalance threshold. The overall objective for the participant is to reduce the level of the graph (lower indicates less nasal) during speech. See-Scape™ provides visual feedback about the amount of air escaping down the nose during speech. If air flows into the tube through the nosepiece placed in the nostril a coloured polystyrene float rises in the tube. The overall objective for the participant is to limit or stop the float rising in the tube during speech. The nasal mirror is placed under the nose and detects nasal escape of air during speech, indicated by fogging of the mirror. The overall objective for the participant is to reduce or eliminate fogging of the mirror during speech.

Figure 10.4.1(a) Nasometry (Nasometer™ II model 6400)

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Figure 10.4.1(b) See-Scape™

Figure 10.4.1(c) Nasal mirror

Biofeedback in VFTh The use of visual biofeedback in this way allows covert physiological processes to be made more overt (Huang et al., 2006), so that the participant can actually see the result of excessive nasal airflow or resonance in a concrete visual form,

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see Figure 10.4.2. Huang et al. also propose that biofeedback serves to heighten sensory cues and also to allow adaptive strategies. In addition, Brunner et al. (2005) assert that visual information provides a strong stimulus to change existing speech patterns.

Performance feedback in VFTh Performance feedback is used in several ways, primarily to support the participant in making sense of the visual feedback display by checking concepts and revising goals. In addition, descriptive terms are used to label the visual feedback display, e.g., ‘towers’, ‘chimneys’, ‘tall/short’, or ‘invisible sounds’. These descriptions may be generated by the participant himself and then developed and reinforced in subsequent sessions. The rationale for incorporating performance feedback alongside biofeedback in VFTh relates to the goals of the intervention and also the feedback tools used. In the motor learning literature, performance feedback is a term used to describe feedback related to the nature of the movement of interest (Schmidt & Lee, 2005). This feedback may be generated by the individual himself or

Figure 10.4.2 Example of biofeedback display with horizontal line showing threshold for individual to monitor and modify nasalance level

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given verbally by another person monitoring the movement. In the context of speech production, therefore, performance feedback can be used by the speech therapist to give immediate and specific verbal feedback about the nature of speech in relation to specified goals. It therefore functions as a conditioned reinforcer of desired speech behaviours whilst also allowing them to be explicitly described, or ‘externalised’, and shaped. In addition, Hooper (2004) comments that most voice therapy programmes use insight and discussion and include a focus on the self-perception of the ‘good voice’; the role played by performance feedback in VFTh. This is true of the majority of speech therapy approaches using biofeedback as explanations and monitoring are provided alongside the visual biofeedback component. In fact, Pantelemidou et al. (2003) argue that this is the aspect that differentiates an approach such as electropalatography from traditional auditory speech therapy, as the individual takes an ‘active’ role in discovering the relationship between tongue placement and speech sound production. In the same way, a feedback treatment for nasal speech could allow someone to actively explore their ability to modify their voice quality in response to visual and performance feedback.

Development of a feedback intervention for nasal speech: Content and phases VFTh uses speech tasks for maximum relevance and the participant progresses in a graded way through the tasks from practice of single speech sounds to sound combinations, single words, word combinations and finally sentences. This sequence represents a hierarchy of complexity as described by Albery and Russell (2005) and which is routinely used in speech therapy. Preliminary target selection is based on initial speech measures and progression and then individually tailored to the participant. VFTh does, however, consist of a number of phases commonly employed in speech and voice therapy programmes (Hooper, 2004), such as: • General awareness of speech behaviour – in this case oral versus nasal voice quality and airflow • Specific awareness of speech behaviour to change – e.g., production of target sounds without nasal emission or nasal resonance • Direct speech production activities – repetitive practice of target sounds and words • Generalisation activities – e.g., conversational speech.

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How might it work? A model for Visual Feedback Therapy Feedback and motor learning models indicate that the speech production system is sensitive to external influences, whilst acoustic speech models offer an insight into the physical properties of speech sounds and the effect of small changes in the system, suggesting that changes in the one aspect of the speech mechanism may have an effect on another. In addition, psycholinguistic theory offers a connectionist perspective of the speech mechanism as a whole, from perception to production. Stackhouse and Wells’ (2001, 1997) speech processing model allows for consideration of the speaker’s stored ‘lexical representations’ of sounds and sound properties and the possibility that these can be replaced with different representations. A model of how VFTh could work in this context is presented below. The model is circular, reflecting the central role of feedback in the intervention, and the contributions of the nasal speaker and the speech therapist are included. There are three core levels leading to the generation of feedback and subsequent speech modification attempts: • The first level is Capacity: for the nasal speaker this refers to the potential to use less nasal speech and includes both physiological potential, i.e., motor skills, as well as learned voice settings. Correspondingly, for the speech therapist this level refers to knowledge of the mechanisms of speech production and behaviour modification. • The second level is Learning: at this level the speech therapist makes explicit the desired speech behaviours and associated mechanisms for change. The nasal speaker can then explore how to achieve these desired behaviours. • The Comparator level describes how the biofeedback tools and performance feedback measure or identify discrepancies in speech behaviours. This is followed by speech modification attempts by the nasal speaker with self-monitoring, supported by the visual feedback generated and contiguous reinforcement and shaping from the speech therapist.

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Conclusion There is sufficient evidence to suggest that a biofeedback intervention such as VFTh could be a treatment option for some individuals with nasal speech, but further research is required. This is clinically important as the current emphasis in the management of nasal speech is often on surgery. Some individuals with nasal speech clearly require surgery as an option, usually where velopharyngeal closure for speech is never achieved (Sell & Grunwell,

Figure 10.4.3 A model of Visual Feedback Therapy

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2001), whilst others with milder or variable symptoms may benefit from diagnostic, and potentially therapeutic, intervention using VFTh. A time limited, focused period of intervention using VFTh could determine whether nasal speech symptoms are modifiable at all and, if so, to a degree acceptable to the speaker and listener. In this respect, the question of what constitutes a clinically worthwhile benefit also requires further examination. Finally, the experience of this author also suggests that nasometry could be more widely used as a therapeutic as well as an assessment tool. The equipment is available at most regional cleft palate centres, is easy to set up and use, and the graphics and games are visually appealing. The availability of portable equipment for use with a laptop computer also means that access to this type of therapy could potentially be extended beyond the cleft centre clinic for wider use in the community with selected individuals.

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Sell, D.A. & Grunwell, P. (2001). Speech assessment and therapy. In A.C.H. Watson, D.A. Sell & P. Grunwell (Eds), Management of Cleft Lip and Palate. London: Whurr Publishers. Sell, D., Britton, L., Hayden, C., Phippen, G. & Russell, J. (2008). Speech and language therapy and nasendoscopy for patients with velopharyngeal dysfunction position paper. Royal College of Speech and Language Therapists. Shprintzen, R.J. & Marrinan, E. (2009). Velopharyngeal insufficiency: Diagnosis and management. Current Opinion in Otolaryngololy & Head and Neck Surgery, 17(4), 302–307. Stackhouse, J., Wells, B. & Snowling, M.J. (1997). Children’s Speech and Literacy Difficulties 1: A Psycholinguistic Framework. London: Whurr Publishers. Stackhouse, J. & Wells, B. (2001). Children’s Speech and Literacy Difficulties 2: Identification and Intervention. London: Whurr Publishers. Tomes, L., Kuehn, D.P. & Peterson-Falzone, S.J. (1997). Behavioral treatments of velopharyngeal impairment. In K. Bzoch (Ed.), Communicative Disorders Related to Cleft Lip and Palate, 4th Edn. Austin, TX: Pro-Ed. Witt, P.D., Rozelle, A.A., Marsh, J.L., Marty-Grames, L., Muntz, H., Gay, W.D. & Pilgram, T.K. (1995). Do palatal lift prostheses stimulate velopharyngeal neuromuscular activity? The Cleft Palate Craniofacial Journal, 32(6), 469–475. Ysunza, A., Pamplona, M., Femat, T., Mayer, I. & Velasco-Garcia, M. (1997). Videonasopharyngoscopy as an instrument for visual biofeedback during speech in cleft palate patients. International Journal of Pediatric Otorhinolaryngology, 41(3), 291–298.

Further Reading Stackhouse, J., Wells, B., Pascoe, M. & Rees, R. (2002). From phonological therapy to phonological awareness. Seminars in Speech and Language, 23(1), 23–34. Sweeney, T. & Sell, D. (2008). Relationship between perceptual ratings of nasality and nasometry in children/adolescents with cleft palate and/or velopharyngeal dysfunction. International Journal of Language & Communication Disorders, 43(3), 265–282.

11 Working in Partnership Catherine Catterall and Sandra Treslove

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Aim of this chapter The aim of this chapter is to enable community-based speech and language therapists (SLT) working with children with cleft palate or velopharyngeal dysfunction (VPD) to offer effective support to the children, their parents and staff working in educational settings. This will be achieved by: • Enabling SLTs to confidently plan therapy, taking planned interventions and surgical procedures into account. • Recommending relationships.

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• Discussing resources and approaches which can be incorporated into therapy sessions or speech programmes in school and preschool settings. • Exploring transition through the education system in relation to the child with cleft palate/VPD.

Introduction Professionals working within regional cleft centres provide early medical care of a child born with a cleft palate. Once primary palate surgery is completed, the child’s care is increasingly provided in community settings by professionals who are not specialists in cleft care. The child will continue to be involved with the multidisciplinary cleft team throughout their pre-school and school years but will be seen in a variety of health and educational settings nearer to home for follow-up of general development, speech and hearing needs. The community SLT will need to be aware of the timing and nature of investigations for orthodontic or surgical treatment when planning therapy. Cleft centre SLTs will have spoken to parents about the close links and liaison they have with community SLT teams working in their local areas. However, it is not uncommon for community SLTs to lack confidence when supporting children with cleft palate on their caseload. Many children with a cleft palate or speech affected by VPD will have speech or language needs in common with their classmates, and the same principles of therapy will apply for both of these groups. However, the emphasis

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of this chapter is on the ways in which the process can differ for children with a cleft palate/VPD, and the issues that SLTs may encounter when supporting them in pre-schools and schools.

Working with pre-schools Many children in the UK will attend a nursery or pre-school prior to starting school. All Early Years providers of childcare must now meet the standards of the Early Years Foundation Stage (EYFS)(Department for Education, 2012a). The framework lays down standards for learning, care and development of children from birth to age 5. It also emphasises the need for a working partnership between professionals involved in the child’s care and the child’s parents/guardians. Service models and protocols vary, but it is likely that for the majority of children with cleft palate, any direct speech therapy intervention prior to starting school will take place in the home or clinic setting. However, it can still be beneficial for SLTs to be in contact with the child’s pre-school or nursery. In such cases, the focus of this additional SLT support is likely to be on ensuring that Early Years Practitioners (EYPs) feel confident about meeting the needs of a child with a cleft palate in their setting, particularly with regards to Personal, Social and Emotional development (PSE) and Communication, Language and Literacy (CLL).

• Note Key recommendations 1. Ensure the setting has an understanding of cleft lip and palate and the impact it may have on a child’s speech, language and communication development. 2. Establish positive links between parents, Early Years Practitioners and the SLT. 3. Recommend activities for the child which can be incorporated into the whole setting.

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How can we work in partnership with Early Years Practitioners to support the child with a cleft palate in the Early Years setting? Ensure that they have a good understanding of cleft lip and palate by providing information about: • Aetiology: Cleft lip and/or palate occurs in early pregnancy when the developing structures do not fuse together correctly. The causes remain largely unknown, although research continues to establish genetic and environmental factors which may contribute (Rahimov et al., 2012; Calzolari et al., 2004; Christiansen, 1999) • Incidence: Cleft lip and/or palate affects approximately 1 in 700 of the population (Vieira, 2008) • Types of cleft: Provide information about the different types of cleft (see Resource CD). Many people are unaware that a cleft lip can occur by itself or alongside a cleft palate, or that it is possible to have a cleft palate without a cleft lip • The role of the palate in speech, and the difference between oral and nasal sounds • The types of speech errors that children with cleft palate may have, e.g., use of nasal consonants, weak sounding consonants and air escaping down the nose accompanying or replacing sounds. Explain that speech errors may be due to the mislearning of how sounds are produced, or may be the result of the palate not functioning correctly • The difference between hypernasal and hyponasal speech: this will help to clarify when there are reports of a child sounding ‘nasal’ • Hearing loss is prevalent in children with a cleft palate. This is because the muscle of the palate is the same one that pulls on the Eustachian tube during swallowing. The muscle may not pull in the right direction. Consequently, the middle ear does not get enough air into it and can become full of sticky fluid (see Chapter 5) • Increasing awareness of the potential for children with a cleft palate to experience delayed language development. Reassure Early Years Practitioners (EYPs) that whatever the underlying cause

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of delayed language development, the same principles for advice and intervention apply to children with a cleft palate as for any other child. Find out about any schemes to support early language development in the pre-school setting, and ensure that the child has access to these as necessary. t Useful Link: The website of the Cleft Lip and Palate Association (CLAPA) is a useful source of clear, accurate information including diagrams and photographs which can support your explanation. www.clapa.com. The National Deaf Children’s Society has a range of resources that are free to download or order, www.ndcs.org.uk. These include posters and pamphlets on ‘glue ear’ which will be of wider interest to many families in the pre-school setting.

How can parents be involved when working in partnership with Early Years settings? Pre-school settings are required by the EYFS to update parents/carers regularly regarding their child’s progress, learning outcomes, and participation (EYFS, 2012). Where an SLT is supporting a child in an Early Years setting, they should also be involved in this information sharing. Parents can be supported to work in partnership with Early Years Practitioners and SLTs in the following ways: • Establish effective channels of communication. This may be through telephone contact, feedback during home visits or clinic appointments, or by arranging a meeting with parents and key Early Years Practitioners. Such meetings can be used to discuss the child’s progress and explore opportunities for joint planning. If the child has complex needs and is known to a range of professionals, a Team Around the Child (TAC) meeting may be appropriate to help the family access the support that they require, as part of the Common Assessment Framework (CAF) process (DfE, 2012b). • Find out the method by which parents are updated about their child’s progress and learning goals, which is itself a requirement of the EYFS (Paragraph 3.67). Use existing modes of communication to provide parents with additional information. For example, the home/preschool link book can also be used by the SLT to give feedback to parents following a visit to the Early Years setting.

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• Gain permission from parents to share information with Early Years Practitioners regarding hearing status and planned medical interventions for their child. • Promote the child’s development of independence skills. It is part of the key person’s role to ensure that every child’s learning and care is tailored to meet their individual needs (EYFS Paragraph 1.11). However, it is important that the child does not become dependent on just one person for their communication needs. To this end, find out how the pre-school will share information so that all staff members feel able to support the child with confidence. This can form part of the training and development needs of the staff, which is a further requirement of the EYFS (Paragraph 1.12).

What activities can be used in Early Years settings to support the speech and language development of the child with a cleft palate? Having provided a pre-school with relevant information as outlined above, it is also helpful to leave them with resources and activity ideas that they can use. The following provides a starting point for activities specifically relating to the needs of a child with a cleft palate.

Sound play ideas Explain that a child will absorb sounds heard repeatedly in their environment into their early speech sound system, and that they will benefit from hearing a wide range of sounds in play situations throughout the day, without being asked to copy them. Try to link ‘sound play’ ideas to daily routines or frequently-played games in the setting, to increase the likelihood of staff adopting the principle (for instance, saying “shhhhhh” whenever turning on taps to wash hands, or including sounds in games such as ‘Simon Says’). Sharing books is a key part of Early Years provision, and provides ample opportunity for sound play activities. Use sound play activities to reinforce the message to Early Years Practitioners not to model sounds with excess tension in the throat. Rather,

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model soft sounds, made with the front of the tongue and the lips. Provide feedback to the children about how the sounds are made, for instance referring to lip shapes or tongue movements.

• Research

A small study (Friberg & Lund, 2010) demonstrated gains in output of speech sounds targeted in joint story telling activities. Although there is limited research evidence available, Psycholinguistic theory would support the recommendation to target the frequency and quality of speech sounds heard by a child, where there is known deficit at the level of output (Stackhouse & Wells, 1997).

Gentle blowing activities When introducing these, explain that the idea is not to improve palate function but rather to encourage children to use an oral airstream. It can be useful to introduce the vocabulary of ‘mouth air’ and ‘nose air’.  Resource CD: For recommended activities and songs, refer to the Oral Airstream Activities on the Resource CD.

Auditory discrimination and awareness games Most SLTs will have access to a wide range of auditory discrimination and awareness games to recommend to pre-schools, so these have not been replicated here.

• Top Tip Listening Walk: This is a lovely activity where children go on a walk to listen for different sounds. The extra dimension is wearing large ears. Go to http:// www.letters-and-sounds.com/resources/p1ears.pdf for a template. When the children return, see if they can recreate the sounds they have heard.

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Link to themes at pre-school Pre-schools usually change their activities and artwork to link in with a particular theme or season of the year. It can be worth exploring ways in which speech activities can be linked in to this. For example: • Life cycles: Why not introduce the dandelion? It follows an interesting cycle as well as the opportunity to gently blow seeds. There are lots of ideas online for pre-school art activities using dandelions. • Seasons: Christmas decorations are often light enough to be blown using a range of speech sounds, e.g., /pʰ pʰ pʰ/, /ʃ/. In the autumn, collect dried leaves and string them up to make a mobile. • Firework night: Encourage the children to think of the sounds that they might hear on firework night, then make a ‘sound picture’ for the wall. Recreate the noise of crackling fire, whistling fireworks, wind in the trees and owls hooting.

Talking stickers A useful means of encouraging a child to talk about what has happened to them in pre-school is through use of ‘Talking Stickers’. The key person writes a brief message on a sticker which the child wears home, describing something that has happened in the day. For example, the sticker could read “We looked at tadpoles today!” This can provide a starting point for conversation, as well as providing a context for families about what a child with intelligibility difficulties might be trying to tell them.

Working with schools The aim of cleft palate care in the UK is for children born with a cleft palate to have normal speech prior to starting school (Royal College of Speech & Language Therapists [RCSLT], 2005). In reality, many children will continue to require therapy input after they start school. Many of the recommendations for supporting pre-school children will still apply. However, for the school-aged child, the RCSLT (2005, p.25) recommends school-based intervention as the preferred model, to promote a collaborative approach to supporting the child. This type of intervention frequently uses a ‘Consultancy Model’, where the child is assessed by the SLT and intervention is then delivered by a member of school

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staff, often a teaching assistant (TA), who follows a programme produced by the SLT (Mecrow et al., 2010). This is often described as indirect therapy.

Supporting the child with cleft palate using a Consultancy Model Although the term ‘Consultancy Model’ is frequently used when describing the way that SLTs work in schools, there is actually little consistency in the way that this model is delivered. There is marked variation reported in the frequency and nature of the input received by the child directly from the SLT, and the way this is followed up by the school (Mecrow et al., 2010). In relation to speech disorders, the RCSLT (2006, p.373) recognises that while therapy should be delivered by a suitably experienced SLT, children can be supported with follow-up practice by a TA, as long as the TA receives sufficient and regular support in modelling of therapy techniques. Video therapy can also be used in place of some face-to-face contact with the SLT. Even TAs who have experience of working with children with speech difficulties may be anxious about whether they will be able to successfully help a child with speech difficulties relating to a cleft palate. This is because different techniques may be suggested to those they have used when working with children with non-cleft speech difficulties (RCSLT, 2005). In addition, the child is likely to present with cleft speech characteristics (CSCs) that they have not encountered before.

• Note Key recommendations 1. Ensure that TAs are able to develop a clear understanding of the rationale for therapy and treatment methods being used. 2. Continue to work in partnership with parents by actively involving them in the therapy process. 3. Work with the child, their parents and educational staff to plan for successful transition throughout the child’s education by considering the transfer of information.

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How can we successfully work in partnership with TAs when supporting the child with a cleft palate? The TA will frequently be the main person to have regular contact with the visiting SLT due to the teaching commitments of the class teacher and special educational needs coordinator (SENCo). Wherever possible, the SLT should meet with all members of staff working with the child, to discuss the child’s needs and therapy input in order to encourage open dialogue should any areas of concern arise. The following areas are worthy of consideration in order to maximise the success of a consultancy or indirect therapy approach: • Use a flexible approach to the delivery of therapy. SLT visits may need to be scheduled more frequently at the beginning of the intervention to give the opportunity for the TA to observe therapy techniques and for the SLT to monitor how the follow-up work is being delivered. The TA can then continue with the follow-up when he/she is confident with the techniques being used and the frequency of therapy is reduced. • Provide clear explanations about the rationale for therapy. Time spent giving information, at an appropriate theoretical level, about a cleft palate/VPD and the resulting speech difficulties will help the TA to develop a strong foundation on which to base the therapy work. School staff have conflicting demands on their time; if they understand that therapy may be part of a diagnostic process (e.g., to determine if the child needs further surgery), they will usually prioritise this work. • Support TAs in developing an understanding of the components of speech. Children have been found to benefit more from comments and feedback given by qualified SLTs in comparison with unqualified people who support children with follow-up practice (Gardner, 2006). Offer the TA specific training on giving the child verbal feedback. This will help them to respond in a more effective way as the child will have accurate information about how their attempt differs from the target. • Encourage TAs to contribute to therapy planning using their knowledge of the child’s interests, strengths and difficulties. Therapy

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activities based on a theme which the child is known to like will be motivating for the child and promote follow-up practice. Similarly, a TA may advise the SLT to avoid using activities which require additional skills such as reading or drawing if they are aware that the child is experiencing difficulties in these areas. • Encourage TAs to use their own knowledge and skills to develop new therapy activities. For example, a TA who has training and expertise in the use of IT may be able to adapt software and applications to motivate the child. • Establish effective channels of communication with the TA. Ensure that the TA is able to contact you with queries between therapy sessions by telephone, email or video-conferencing (where available). During face-to-face contact, encourage the TA to give honest feedback about practice carried out. Carry out joint problem solving as part of the therapy sessions to review any activities that have been judged as unsuccessful by the TA. • Include successful classroom based strategies into the Speech and Language Therapy input. This can include: • Visual support materials used to help the child’s attention and listening skills, behaviour and understanding of routine and transitions • Communication strategies such as the use of computer-generated symbols to support the language component of activities that enable the child to focus on their speech production • Reward schemes. • Share ideas for therapy resources. TAs may have limited time for producing resources to use in follow-up practice sessions and will benefit from advice on how to develop a small set of games or activities that can be used to support therapy for a range of sounds and levels. This will enable the TA to spend more time with the child and less time on the production of resources. Again, a TA who understands the underpinning theory and rationale for therapy will be able to adapt existing games and therapy materials more effectively.

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How can we involve parents in school-based therapy? Parents can continue to play an active role in their child’s therapy when their child starts school by attending therapy sessions in school. This will enable them to maintain contact with the SLT, learn about therapy techniques being used, and plan ways in which the child can continue to be supported with their therapy practice at home. Where parents are able to attend therapy sessions in school alongside a member of school staff such as a TA, it may be helpful to consider the following issues: • Some children find it difficult to be separated from their parents part way through the school day. It may be appropriate to schedule the therapy appointment for the beginning or end of the school day to help the child to cope with this. • Some children find it confusing to have their parent take part in a ‘school’ activity. Parents and school staff should discuss who will take the lead role during the session to ensure that the child knows and understands what is expected. • Some children may perform better in school when their parents are not present. In this case, it may be better for parents to meet with the SLT and TA after the therapy session or to attend occasional sessions to observe the therapy activities and discuss their child’s progress. • Parents and school staff should decide who will take a lead role in carrying out follow-up practice with the child. A method of communication should be agreed between parents and the school to record practice carried out and the child’s progress. Parents may find it preferable to support the children with small homework tasks such as practising ‘word of the day/week’, which can be carried out alongside other homework tasks. • Reward systems for speech practice should be made consistent between home and school. • Older children with persisting speech difficulties are likely to be less receptive to support from their parents for speech difficulties than younger children with phonological delay (Pascoe, Stackhouse & Wells, 2006).

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If parents are unable to attend therapy sessions in school it is possible to make a formal arrangement with parents about how information will be shared. Parents should have a clear idea of how often they can expect to receive feedback and how this will be received: • Choose a mode of communication that the parent is comfortable with. Telephone, email, SMS text messages and handwritten communication can all be used if local policy allows and if parents are in agreement. • Use existing modes of communication such as home–school link books to provide feedback for parents where possible. Information shared in this way can then also be read by teaching staff. • Ensure that communication is a two-way process between parents and the SLT. Issues such as parental literacy levels can impact upon communication between parents and professionals if the most appropriate mode of communication is not chosen. Concerns on the part of the parent about communicating in writing may be misinterpreted as unwillingness to work in partnership with SLTs and school staff. Finally, it is important to help parents to form realistic expectations regarding their child’s progress and the possible outcomes of therapy. Encourage open and honest discussion regarding the therapy process, the therapy techniques being used, and expectations regarding follow-up by school staff and parents. Explain to parents how decisions will be made about discharging their child from Speech and Language Therapy in order to make this as positive an experience as possible when it occurs (Baker, 2010).

How can we work with children, their parents and teachers to ensure that the processes of transition and transfer are a positive experience? Throughout childhood, children go through the continual process of transition as they move from one stage of their education to the next. In particular, the process of transfer between education establishments can lead to anxiety for

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many children and their parents, but for children with a communication difficulty, this can be even more challenging (Ayre & Roulstone, 2009). “The key to a successful transition is the sharing of relevant information” Wade, 2008, p.10 Teaching staff that are working with the child at each stage of transition will need information about a range of issues relating to the child’s cleft palate. They may have limited experience of working with a child who has a cleft palate and need advice on reliable sources of information about the condition. Information should be provided about the nature of the child’s speech difficulties and the way in which these need to be supported through therapy input, follow-up practice, and general support strategies in the classroom. It may be helpful to inform teaching staff about the child’s involvement with the multidisciplinary cleft team and the types of support that they receive from the different professionals. School staff are likely to be familiar with members of the local child health team but may not have previous experience of a child receiving care from a regional team.

• Note Care Pathway At each transition stage, it will be useful to provide information regarding the child’s on-going cleft care for all those involved in educating the child. It may be useful to point out that, although the child would have had surgery to repair their lip and palate in infancy, for many children, multidisciplinary cleft care can involve a 20-year process of monitoring and treatment which may include: • Routine multidisciplinary reviews at the regional cleft centre at the ages of 5, 10 and 15 • Regular audiology reviews (up until the age of at least 7 and possibly beyond) • Further surgery if the function of the palate is still not adequate for clear speech. The timing of this will vary from child to child

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• Orthodontic treatment (required because the presence of a cleft can significantly disrupt the growth and alignment of teeth) • Alveolar bone graft surgery between the ages of 8 and 11 for children with a cleft involving the alveolus (gum) • Lip, nose or jaw surgery in late teenage/early adulthood, if desired at that stage by the individual. There may be concern about children missing school while they attend appointments, or have further surgical treatment. This can be addressed by advising school staff of the frequency and nature of the appointments and keeping them informed of the outcome and any planned treatment.

• Note Although at each stage the primary focus of therapy input will be helping the child to develop their speech sound system, it is essential that systems are in place to enable the child to express themselves effectively in the short or long term, as required. These systems could include: • Sign-supporting systems – e.g., Signalong© www.signalong.org.uk and Makaton®; www.makaton.org • Communication books – for further information see Developing and Using a Communication Book by Latham; www.ace-centre. org.uk • High-tech communication aids – for further information, see www. communicationmatters.org.uk/page/high-tech-aac • Communication applications for smart-phones and tablet devices can provide an alternative to some high-tech communication aids and may be suitable for children who are verbal, but need some help in getting their message across clearly.

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How can information be shared during periods of transition and transfer? All of the usual methods of sharing information are appropriate for use with a child with speech difficulties relating to a cleft palate. These include formal written reports and transition meetings. For younger children, this transfer of information will primarily take place without their involvement, with parents, healthcare professionals and Early Years Practitioners providing written and verbal information for school staff. However, for the older child with more persisting speech difficulties, it becomes increasingly important for them to be involved in the transfer of information. This is because: • Older children are likely to have developed opinions on the types of strategies that they find helpful and how they would like this information to be shared • As children progress through the education system, they will come into contact with a wider range of teaching staff • The child with a persisting speech difficulty may be concerned that people notice their speech difficulty before their abilities and personality. Child-led transfer of information allows the child to communicate about their strengths and interests as well as areas that they find difficult.

• Note

Transfer of information: Involving the child The following documents can be produced by parents, educational staff and the child to provide key pieces of information: Communication passport – A document containing information about the child, including medical needs, areas of strength and difficulty, and ways in which other people can facilitate

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communication with the individual (Millar & Aitken, 2003). See www.communicationpassports.org.uk for further information and templates which can be fully adapted to meet the needs of the individual communication passport user. One page profile – A single page document which provides key pieces of information about the child’s strengths, difficulties and ways of supporting them (Erwin & Sanderson, 2010). A copy of this document can be placed in the class register or distributed to members of teaching staff. Communication cards – Small cards giving basic information about the child’s needs and useful strategies. These can be introduced if there is a breakdown in communication caused by the child’s speech difficulties.

Conclusion A significant number of children with cleft palate and/or VPD will require therapy input in order to achieve the best speech outcomes. Successful partnerships between SLTs, parents, educational staff and the children themselves, established during the Early Years Foundation Stage and continued throughout primary and secondary school, will allow for therapy to be effectively integrated into their education. The SLT will play an important role in ensuring that education staff have adequate information regarding the child’s condition where it impacts on speech and language development. They will advise on therapy techniques specific to the treatment of palate-related speech difficulties, which may or may not differ from those routinely used in these settings to support children with speech and language difficulties. The complex speech difficulties which can be associated with cleft palate and VPD can be daunting for even the most experienced SLT. However, it is hoped that the recommendations and resources presented in this chapter, combined with the community SLT’s existing expertise in supporting children with a wide range of speech, language and communication difficulties in educational settings, will result in the child with a cleft palate receiving effective support at any age and stage of their education.

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• Questions Frequently Asked by Educational Staff

Why is there more than one SLT involved?

Staff in educational settings, as well as families themselves, may be unclear about the different role of the SLT from the community base and that of the SLT from the regional cleft centre. It can be helpful to establish this from the start.

Can we introduce phonics for a child with a cleft even if their speech is unclear?

Explain that there is no reason to exclude children with a cleft from phonics teaching – the listening activities in particular can be really helpful. Ensure that staff know to model all sounds gently. If a child is attempting to make the sounds and is getting frustrated and/or saying them incorrectly, suggest that staff encourage them to listen and watch instead. Suggested phrases include, “Just use your ears now, your mouth is having a little rest”. A child who is not able to say sounds can show they understand the link between sound and letter by pointing to what they hear.

What should I do if the child pronounces a word incorrectly? Be sure that all in the setting are familiar with the concept of modelling, rather than correcting errors. Advise that the child needs time to learn by listening, watching and seeing. Recommend that a word is repeated back to the child in a natural context, e.g., “Yes, it’s a cat, a big cat” without any demand being made on the child to repeat the word.

What do I do if other children comment on the child’s cleft lip or on the fact that they sound or look different?

Young children are extremely interested in everything around them. As part of this natural curiosity, they may ask questions about, comment, or even reach out to touch the face of a child with a visible difference like a cleft lip. As adults, we sometimes struggle in knowing how to respond to this interest. Ignoring the fact that a child may look or sound different will not satisfy their peers’ curiosity, and may lead to the child feeling that their cleft is something that should not be talked about. The best approach is to simply answer any question in an age appropriate way. For example:

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“Jack was born with a gap in his mouth which was closed when he was a baby.” “Jack has a scar but it doesn’t hurt him.” You could then emphasise what they have in common with Jack, e.g., “Jack really likes playing with the blocks too, shall we play together?” “Has anyone else ever been in hospital?” Even in older children, comments and questions may not be meant unkindly and it may help a child to develop strategies to respond positively. However, issues of bullying may arise and must be dealt with effectively by the school. Ensure that school staff, parents and the children know that they can contact the clinical psychologist from the cleft team for further advice.

References Ayre, A. & Roulstone, S. (2009). Transition to Secondary School: Supporting Pupils with Speech, Language and Communication Needs. Report to the Communication Trust. [Electronic version] www.thecommunicationtrust.org.uk. Retrieved September 18, 2012 from https://www.thecommunicationtrust.org.uk/media/13562/transition_report_final. pdf Baker, E. (2010). The experience of discharging children from phonological therapy. [Electronic version] International Journal of Speech-Language Pathology, 12(4), 325–328. Calzolari, E., Bianchi, F., Rubini, M., Ritvanen, A. & Neville, A. (2004). Epidemiology of cleft palate in Europe. Cleft Palate-Craniofacial Journal, 41(3), 244–249. Christiansen, K. (1999). The 20th century Danish facial cleft population – Epidemiological and genetic-epidemiological studies. Cleft Palate-Craniofacial Journal, 36(2), 96–104. Department for Education (2012a). Early Years Foundation Stage. Available from https://media.education.gov.uk/assets/files/pdf/e/eyfs%20statutory%20framework%20 march%202012.pdf Department for Education (2012b). Team Around the Child (TAC). Available from http://www.education.gov.uk/childrenandyoungpeople/strategy/integratedworking/ a0068944/team-around-the-child-tac Erwin, L. & Sanderson, H. (2010). One-page profiles with children and young people. [Electronic version] Helen Sanderson Associates. Retrieved May 1, 2013 from http:// www.helensandersonassociates.co.uk/ media/35085/onepageprofilesinschools.pdf

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Friberg, J.C. & Lund, K.K. (2010). The effects of supplemental joint storybook reading on preschool students use of strident sounds: A preliminary investigation. Issues In Communication Science and Disorders, 37, 174–180. Gardner, H. (2006). Training others in the art of therapy for speech sound disorders: An interactional approach. [Electronic version] Child Language Teaching and Therapy, 22(1), 27–47. Mecrow, C., Beckwith, J. & Klee, T. (2010). An exploratory trial of the effectiveness of an enhanced consultative approach to delivering speech and language intervention in schools. International Journal of Language & Communication Disorders, 45(3), 354–367. Millar, S. & Aitken, S. (2003). Personal Communication Passports: Guidelines for Good Practice. Edinburgh: CALL Centre, University of Edinburgh. Pascoe, M., Stackhouse, J. & Wells, B. (2006). Persisting Speech Difficulties in Children: Children’s Speech and Literacy Difficulties, Book 3. Chichester: John Wiley & Sons Ltd. Rahimov, F., Jugessur, A. & Murray, J. (2012). Genetics of nonsyndromic orofacial clefts. Cleft Palate-Craniofacial Journal, 49(1), 73–91. Royal College of Speech and Language Therapists (2005). Clinical Guidelines, 3rd Edn. [Electronic vversion] Bicester: Speechmark Publishing Ltd. Retrieved September 18, 2012 from http://www.rcslt.org/members/publications/clinicalguidelines Royal College of Speech and Language Therapists (2006). Communicating Quality 3 – RCSLT’s Guidance on Best Practice in Service Organisation and Provision. London: RCSLT. Stackhouse, J. & Wells, B. (1997). Children’s Speech and Literacy Difficulties: A Psycholinguistic Framework. London: Whurr Publishers. Vieira, A.R. (2008). Unravelling human cleft lip and palate research. Journal of Dental Research 87, 119–125. Wade, C. (2008). Video profiling: Smoothing school transition. RCSLT Bulletin, 680, 10.

12 Working with Children with Complex Needs: Meeting the Challenges Zoe Gordon and Carrie Luscombe

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“Prob...lem...sol...ving?” Pooh said slowly, scratching his head. “That sounds like a ‘what’. The ‘whats’ are easy. It’s the ‘hows’ that are difficult.” (From: Inner book jacket, Winnie The Pooh on Management and Problem Solving by Roger Allen, 1995)

Aim of this chapter This chapter aims to: • Discuss the key notions for working with children with complex needs, as outlined in current national guidelines and research literature, and what this means in practice for clinicians, children and families. • Present case studies to highlight the challenges the clinician may encounter when working with children with cleft and complex needs and suggestions for how these can be addressed. • Provide information on cleft-related syndromes that clinicians are most likely to encounter in community practice. • Signpost clinicians towards outcome measures for children with complex needs.

Introduction Definitions of ‘complex needs’ can be diverse and difficult to operationalise. The complexity of care needs for children in the community has increased rapidly over the past decade due to advancements in medical care. In this chapter we will be examining the needs of children with cleft-related speech, language, communication or feeding difficulties in the presence of additional sensory, physical, developmental, or emotional-behavioural needs. Each child presents with a unique interaction between these different needs, even if the recognised needs are not necessarily severe. As discussed in previous chapters, the baby born with a cleft faces a longterm journey through well-established surgical and therapeutic interventions. For some children with a cleft plus other associated anomalies there will be additional challenges for the SLT, as the child may not fit into the routine pathway

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of service provision for a child with a cleft. As speech, communication and feeding difficulties can be important manifestations of a range of congenital conditions, related to both cleft and non-cleft conditions, the SLT can be key in recognising syndromic features in a young child and bringing this to the attention of the multidisciplinary team (Reilly, 2006). There are over 400 documented syndromes associated with cleft (Wyszynski et al., 2006; Winter & Baraister, 2001) and prevalance rates of syndromic cleft palate in the literature vary from 15% to 60% (Juggesur et al., 2009; Eppley et al., 2005; Stoll et al., 2000; Schutte & Murray, 1999) depending on the geographical area of the study group, ethnicity, variability in the definition and expression of a syndrome, and changes in prevalence over the past 20 years. It is recognised, however, that there is a higher incidence of additional medical and psychosocial anomalies in children with isolated cleft palate than in those with cleft lip and palate, or with cleft lip only (Mossey & Modell, 2012; Wyszynski et al., 2006; Stanier & Moore, 2004; Schutte & Murray, 1999).

Working with children with complex needs: Notions and guidelines Since the turn of this millennium there has been an increased commitment to improved service delivery for children with complex needs, building on the principles set out in the UN Convention of the Rights of the Child (1989). A number of strategic documents have been instrumental in shaping services (see Table 12.1). The term ‘complex needs’ naturally guides service delivery towards a model of collaboration between health, education and social care (Rankin & Regan, 2004). In this context, the Specialised Services National Definitions Set 15 (SSNDS, 2010) calls for cleft teams to provide an integrated service of high-quality care and support to children and their families. “Coming together is a beginning; keeping together is progress; working together is success.” Henry Ford Foremost in clinical practice guidance is an emphasis on improving holistic, multidisciplinary, integrated team working. This requires information sharing across the agencies and it is vital that team member roles are clearly defined to enable families to navigate services appropriately. In practice, strained

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Table 12.1 Timeline of key policies shaping services for children with complex needs

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resources and the evolving needs of families mean this can be challenging to implement. There is a range of national and local initiatives striving to formalise this. The Department for Education guidance in the form of The Common Assessment Framework, ‘Team Around the Child’ (TAC) meetings and Early Support have sought to build more responsive, coordinated services. SLTs are often key members in these processes, particularly for children with more complex communication needs. g Further Reading: http://www.education.gov.uk/childrenandyoungpeople/ strategy/integratedworking/caf/ a0068957/the-caf-process Table 12.2 When is there time for our family? An excerpt from Alfie’s mum’s diary Mon

Tues

Weds

Thurs

Fri

6th 7th am: GP Nursery (Immunizations) 2pm Portage

8th

9th 2.30pm Community nursing team home visit

10th am – Playgroup

13th

14th Local ENT clinic

15th

17th am – Playgroup

20th am: GP 2pm Portage

21st Nursery

22nd Cleft specialist nurse & SLT

16th 11am Community nursing team home visit 23rd Team around the child ‘TAC’ meeting

28th 27th Regional hospital Nursery – Cochlear implant team assessment

29th Health Visitor clinic 11am Social worker visit

30th 1st Physio and OT at Neurosurgery local hospital clinic – tertiary hospital

4th 2pm Portage

6th CCN team

7th Regional eye hospital (45 miles from home)

5th 11am Hip scan 3pm Dietitian

24th Respite

8th am – Playgroup pm – Neurology appointment

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Coordinated service provision also helps to ensure that the needs of children and young people are seen within an empowered family context (National Service Framework, 2004). Parents need to be equipped with timely, evidence-based information in order to make informed decisions about their child’s care and have the opportunity to share and receive support from other parents who understand the real experiences of having a child with not only a cleft but additional needs (Carter et al., 2007). Parents of a child with complex needs may have to quickly become specialists in a wide range of areas, juggling the needs of their child, seeing various professionals and keeping appointments, whilst balancing their role as mum or dad. Individual health professionals will at the same time be identifying targets for intervention. Use of therapy outcome plans, health action plans, Individual Education Plans (IEPs), family support meetings and multi-professional planning are commonplace tools to facilitate realistic and achievable target setting. Nevertheless, if multiple professionals are involved it is crucial that targets are not only joined up but also prioritised so not to place an unreasonable burden of care on families. In addition to ensuring joined-up care with a range of other professionals, interdisciplinary SLT liaison is also essential. It is not unusual for a child with a cleft and complex needs to have more than one SLT involved in their care, again adding to the number of professionals for the child and families to engage with. Examples of when joint SLT working is often required: • A child with congenital muscle weakness, due to a neurological problem, also has a cleft palate. As the child develops, both community, specialist and cleft SLT expertise may be required to determine whether speech difficulties are cleft/VPD-related or whether the presenting communication difficulty is more likely to be associated with the child’s other neuromotor challenges • A child who has a moderate sensorineural hearing loss and a cleft palate • A child who presents with dysarthria and/or sensorimotor planning difficulties and a cleft. In these circumstances it is often helpful for a lead SLT to be agreed between the therapists and carers as the main contact and coordinating link. This therapist may change over time according to the child’s needs.

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g Further Reading: Gascoigne, M. (2006). Supporting Children with Speech, Language and Communication Needs Within Integrated Children’s Services. Position Paper. RCSLT: London.

Working with parents and families: Learning from the experts “Having a child with complex health needs in the family is […] a combination of joy at achievements however limited, and sorrow from missed opportunities and distress experienced.” McConkey, Barr & Baxter, 2007, p.12. Despite the wealth of professional expertise available to support children with complex needs it is arguably parents and families who hold the greatest understanding of their child’s needs. It is therefore vital that parents and children,

Figure 12.1 How supportive is support? Juggling specialities within speech and language therapy

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where possible, are engaged as equal and involved partners in decision making and target setting. In addition, clinicians should be mindful of: • Environmental differences • Cultural differences • Psychosocial influences. These factors may individually or in combination affect the families’ decision making and priorities. In addition, degrees of parental support can be highly variable although the literature has shown that parents, particularly those of children with more extensive clefts, generally have a high degree of positive coping and outlook (Nelson et al., 2012; Baker et al., 2009).

• Research

MacDonald and Callery (2007) interviewed 26 parents of children with complex needs, and 17 professionals, to undertake one of the few studies looking at changes in child and parental needs over time. The aim of these in-depth interviews was to devise a developmental care map. They found that in infancy the care needs of a child were disguised by the typical dependency associated with infant development. During this stage parents often received the most support from family and friends. However, as children moved towards school age the needs of the child and differences between their age-matched peers became more obvious. They established that at this stage parents were more likely to experience fatigue, depression, isolation, restrictions in returning to work and less support from family and friends. As children grow older family homes can also become increasingly ‘medicalised’ with equipment in the form of wheelchairs, hoists, home adaptations and medical supplies.

What have parents said they want from the clinicians caring for their child? “We’ve met so many professionals I can’t remember all their names or even who does what. I’d never even heard of some of their jobs […] It’s quite amazing that I have you all here to help him but it’s so scary that he needs all this help. I appreciate it but just wish we didn’t need it. Friends from my NCT class

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have all met up but I haven’t been able to see any of them yet as we have so many appointments. I just can’t wait for a normal mums’ coffee morning and some cake!” Mum of Joseph (aged 4 months) Parents have repeatedly identified particular characteristics that are important in clinicians caring for their children with, and without cleft. These include: • Good knowledge in their specialist area • Clear communication • Sensitivity • The ability to convey concern • Continuity of care. Clinicians who demonstrated these qualities have been perceived as more competent and trustworthy (Nelson & Kirk, 2012; Stone et al., 2010). These are similar to findings in the wider non-cleft literature. Research by Carter et al. (2007) identified key themes of: • Sharing • Clinician–parent dialogue • Respect • Trust • Choices • Flexibility. In practice, parents frequently comment on their battles to access support services and to then manage the volume of information and advice they are given. Qualitative research examining parental needs has suggested some improvements in how clinicians can deliver information. Nelson and Kirk (2012) highlighted parents’ needs for regular updates, and individualised rather than generic care plans for their children with a cleft. It was felt that tailored support helped parents and children prepare for the potential timing of future interventions, and particularly in the planning for transition from children’s to adult services.

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• Research

A review by the Local Authority Research Consortium (Easton et al., 2012) recognised that families can initially be fearful of formalised processes such as the Common Assessment Framework (CAF) due to lack of information sharing around the purpose of multidisciplinary assessment. However, the review found that all families interviewed could identify at least one area of improvement in their family situation following the CAF process. It has been recognised that some universal services require an increased understanding of when families could be better supported through the CAF process (Munro, 2011). g Further Reading: Norgate, R., Traill, M. & Osbourne, C. (2009). Common Assessment Framework (CAF) – Early views and issues. Educational Psychology in Practice, 25(2), 139–150.

Challenge No. 1: Cleft surgery – now, later, or not at all? The debate on the optimal age for cleft palate repair is ongoing. At most UK cleft centres the aim is to repair the child’s cleft palate prior to 12 months of age. For some children with complex needs, particularly those with respiratory or cardiac difficulties, this may be delayed for several months. This is generally to ensure that priority surgeries/assessments can be undertaken (e.g., heart surgery, neurosurgical shunt insertions, gastrostomy placement, sleep studies) and to guarantee that the child is medically stable enough for surgery. Children with particularly hypoplastic palates or mandibles, i.e., those with Pierre Robin Sequence, Treacher Collins syndrome, 22q11 deletion syndrome, Goldenhar syndrome, or hemifacial microsomia, can also be technically challenging for the cleft surgeon to repair when children are very small. This may mean that surgery is delayed for several months to allow for growth. Ethical questions around whether or not to offer surgical treatment to repair a cleft may be raised when managing a child with the most complex of care needs. Such cases may include: • Children with life limiting conditions with expected pre-school mortality

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• Those affected by profound motor and cognitive impairments, e.g., level 5 impairments on the Gross Motor Function Classification System (GMFCS) for children with cerebral palsy who may never develop spoken communication • Children with disabilities who are so unwell that their participation in society may be severely limited. Whilst the designated centres in the UK are specialists in the care of children born with a cleft, community therapists, paediatricians, and allied health professionals contribute crucial information to the discussion around the appropriateness of cleft repair for such children. Discussions may centre around putting a child through an anaesthetic for cleft surgery when the surgical outcome for speech is likely to less favourable. Complicating factors for the initial cleft repair, e.g., a small mandible and reduced mouth opening, can make it almost impossible for the surgeon to do as good a job as they aim for. Furthermore, little muscle/tissue may lead to palatal breakdown and the need for further palate repair, or a repeat procedure when the child has grown more and access is easier. The issue is whether this procedure will have a sufficient impact on the child’s long-term communication ability. This could be debated for the child with neuromotor difficulties and severe learning difficulties. There is often no clear right or wrong in these cases, and professionals may have widely varying opinions. These questions do open up the ethical debate about how best to care for a child whose future is likely to differ from the norm. A judgement of risk is also sometimes required if the cleft surgery may further compromise the child’s airway or health status. Such children need careful multidisciplinary team discussion and their parents careful counselling. g Further Reading: Body, R. & McAllister, L. (2009). Ethics in Speech and Language Therapy. Chichester: John Wiley & Sons Ltd.

• Case study: Ruby

Diagnoses: submucous cleft palate, atrial septal defect in the heart, GMFCS level IV 4 limb cerebral palsy, MRI showing cortical atrophy in fronto-temporal lobes and agenesis of the corpus callosum, recurrent pneumonia. Early history: Ruby was a term baby born at her local hospital and diagnosed

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with a small hole in the heart. Her parents left hospital when she was 3 days old to establish feeding at home. She was re-admitted to hospital, aged 5 days with a chest infection which was treated with antibiotics. Her parents self-discharged from hospital when she was 15 days old and moved to India when she was 17 days old. Upon arrival in India she was re-admitted to hospital with pneumonia and diagnosed with a submucous cleft palate. The family returned to the UK for work when Ruby was 12 months old. By this stage she presented with severe developmental delay, an abnormal MRI and had had six admissions to hospital in India for respiratory infections. Cleft team referral: Ruby was reviewed by the consultant cleft plastic surgeon at age 15 months to discuss repair of her submucous cleft palate. Due to her profound learning difficulties and cardiac problem the consultant decided that it was not in Ruby’s best interests for the palate to be repaired as it would not improve her speech outcome. Ruby was referred to the cleft team dysphagia specialist for assessment of her feeding at 18 months. SLT assessment: Indicated a severe oral-pharyngeal dysphagia with signs of aspiration when drinking thin fluids, and large volumes of nasal regurgitation. Ruby showed clear signs of distress when feeding, particularly when food and fluid came down her nose. Her mother had resorted to feeding her purée by pouring large spoonfuls into her mouth each time she cried in an attempt to maintain her nutrition. Ruby’s mother explained it was easier to feed her lying down as it stopped food coming down her nose. At this time, Ruby was starting to mouth toys and was communicating using vowel sounds and /uma/ but was not attempting any reduplicated babble. Advice was given around optimal food and fluid viscosity for safer feeding and positioning for feeding. Objective assessment: A videofluoroscopy assessment of Ruby’s swallowing demonstrated: - Nasal regurgitation on each mouthful of food due to her unrepaired submucous cleft - Fluid which did not clear as quickly as children without neuromotor difficulties due to muscle incoordination - A delayed swallow but no frank aspiration of food or fluid into the airway

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- Poor clearance of food and fluid from the pharynx placing her at increased risk of aspiration. Next steps: Ruby was referred to the surgical team for consideration of a feeding tube and to the community complex needs SLT team for communication support. Ruby’s parents were eager for her cleft to be repaired in the hope that this would resolve her feeding difficulties and allow her to speak. Ruby’s family required repeated sessions with the SLT and paediatrician to develop their understanding of Ruby’s feeding difficulties. It was explained that repairing the cleft was not in itself going to improve her oral motor difficulties, reduce her aspiration episodes or make her speak. However, the unrepaired cleft was significantly contributing to her oral aversion due to the nasal regurgitation of food. SLT follow up: Ruby was seen by the cleft dysphagia therapist and community complex needs specialist SLT for two joint communication reviews three months later. During the sessions Ruby was engaging in lovely vowel and /mama, nana/ babble turn-taking with her parents and the therapists. She was eye-pointing to indicate her preferences between toys, showing some understanding of family names and her favourite toys and was vocalising to protest or gain an adult’s attention. By this point Ruby was being fed by nasogastric tube and offered small amounts of food which she typically declined. What should we do? Whilst obviously no one could accurately predict what the future held for Ruby, it was felt that she was showing intentional communication and verbal, vocal interaction. This was limited to “mama, nana” due to her inability to produce pressure sounds. By leaving her palate unrepaired was thus limiting the potential for Ruby to develop spoken communication. We were unsure whether further words or consonant sounds could potentially develop. A multidisciplinary team decision: A multidisciplinary review, involving 15 professionals including the two SLTs, paediatrician, dietitian, social worker, physiotherapists, OT, specialist nurses, neurologist and community nurse, was arranged to discuss Ruby’s progress. At this point it was decided to re-refer Ruby to the cleft surgeon for consideration of palate repair, who in turn liaised with the cardiologists to ensure that she was stable enough for surgery.

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Palate repair: Ruby’s palate was repaired aged 2 years 4 months. Within a few months of surgery Ruby was accepting more tastes of foods, smiling and vocalising whilst exploring food, with no nasal regurgitation. Although she was only having small volumes there was a measurable improvement in Ruby’s acceptance of food tastes and also parent–child interaction during social mealtimes. Fifteen months later (aged 3 years 7 months) she still showed a preference for nasal sounds but was able to achieve very weak /g, d/ sounds in her dysarthric vocalisations. She was attempting to imitate some single words and could achieve some /d, g/ sounds in word final position. What does this show us? - Care pathways are essential in structuring services, but each child and their family must be considered individually. - Multiprofessional liaison, joint assessments, debate and discussion are key in achieving the best possible outcomes for families. Liaison does not necessarily mean professionals will all be in agreement about the best management option. - We will frequently need to consider ethical issues in our practice, where both the risks and benefits for a child may be uncertain for years to come.

Personalising our advice Advice on supporting early communication in children born with cleft palate but without complex needs is firmly established in cleft care pathways. Early advice/intervention sessions are frequently offered. However, there will be children who are too unwell or developmentally delayed for a standard early advice session. Whilst the cleft palate or communication development may not be the priority area for parents of a young child with complex needs, these are the children who often require most support. An early advice session to inform parents about the potential impact of the cleft can still be offered, but adapted to reflect the child’s individual needs. Timing of such advice often requires careful judgement on an individual basis for each child and family and sessions may be most effective when delivered jointly between the specialist cleft SLT and the community SLT.

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Challenge No 2: The child with a cleft and tracheostomy In recent years, advances in medical technology and neonatal care have enabled an increased number of children with complex needs to go home from hospital with a tracheostomy. This means that community therapists are now more likely to encounter a child with a tracheostomy on their caseload and be involved in supporting these children through early childhood and into school. Children experiencing breathing difficulties, which cannot be managed through non-invasive ventilation or infant positioning techniques, may require a surgical or percutaneous procedure called a tracheostomy. This enables a child to breathe by creating an opening in the front of the trachea below the level of the larynx. A tracheostomy tube is then inserted in order to maintain the opening, or ‘stoma’. Indications for tracheostomy in children are typically different than those in adults. For children born with a cleft, airway obstruction necessitating tracheostomy is most frequently seen in children with small or retracted jaws which can result in the tongue base obstructing the upper airway. These are key feature of Pierre Robin sequence.

Figure 12.2 Tracheostomy placement

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• Terminology

Micrognathia Retrognathia Glossoptosis

Small jaw Retracted jaw Posterior displacement of the tongue

In addition, children born with a cleft may have co-occurring airway problems resulting from: • Neurological impairment • Craniofacial deformities affecting the airway structure, resulting in obstruction • Laryngeal anomalies, e.g., severe laryngomalacia, adductor vocal fold paralysis, or laryngeal webbing • Lower airway problems such as lung abnormalities, lower airway collapse, severe chronic lung disease

Figure 12.3 Four-week-old child with micrognathia and retrognathia

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• Subglottic stenosis which may be congenital or arise due to longterm ventilation in the neonatal period • Acute airway infection.

• Terminology Laryngomalacia Also known as ‘floppy larynx’, a congenital abnormality/immaturity of the laryngeal cartilage. This can cause the supraglottic structures to collapse into the airway on inspiration, causing airway obstruction. Subglottic stenosis Narrowing of the subglottic airway. Adductor vocal fold paralysis Vocal cords are maintained in a closed (adducted) position. Chronic lung disease Damaged tissue in the lung. In babies and children this is most commonly associated with prematurity. g Further Reading: Seidman, A., Sinz, E. and Goldenberg, D. (Eds) (2011). Tracheotomy Management: A Multi-disciplinary Approach.

The impact of tracheostomy on communication development If we believe early vocalisations and babbling to be the building blocks of later speech production and language development then young children with a tracheostomy are clearly at a disadvantage. If the tracheostomy is ‘tight to fit’ in the trachea (Figure 12.4), air is inhaled and exhaled through the tracheostomy and by-passes the vocal folds and mouth. A ‘tight fit’ is common in neonates and the younger baby due to their small airways. This lack of airflow through the vocal cords will result in aphonia. Aphonia can influence early parent–child interactions as parents do not receive the meaningful crying, giggling and cooing cues from their baby that are so crucial to early socialisation. The aphonic child is unable to experience vocal turn-taking and the auditory feedback of their own babble patterns. Parents may stop talking to their babies when their communication

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Figure 12.4 A tight tracheostomy tube in the trachea

attempts are met with silence and children may be at increased risk of delayed speech and language development if adults do not recognise their non-verbal communication attempts (Bohm et al., 2010; Kraemer et al., 2005; Orringer, 1999; Adamson & Dunbar, 1991).

Does the age of tracheostomy placement affect communication outcomes? The younger child Opinion is divided on whether or not the presence of a tracheostomy impacts on the communication development of a young child with no underlying neurological impairment. A number of reports support the theory that the age at which a tracheostomy is placed is influential on speech and language outcomes. An increased risk of speech and language impairment has been identified if the tracheostomy is placed during the pre-verbal stage of linguistic and phonetic development