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1 CLEFT SPEECH A TELLTALE SIGN OF OCCULT SUBMUCOUS CLEFT: A CASE STUDY MS. ARPITA CHATTERJEE SHAHI AUDIOLOGIST AND SPEECH LANGUAGE PATHOLOGIST ALI YAVAR JUNG NATIONAL INSTITUTE FOR THE HEARING HANDICAPPED EASTERN REGIONAL CENTER, B.T. ROAD, NIOH CAMPUS BONHOOGHLY, KOLKATA MR.INDRANIL CHATTERJEE LECTURER ALI YAVAR JUNG NATIONAL INSTITUTE FOR THE HEARING HANDICAPPED EASTERN REGIONAL CENTER, B.T. ROAD, NIOH CAMPUS BONHOOGHLY, KOLKATA ABSTRACT The following article presents a case study of a child diagnosed with Occult Submucous cleft palate. Submucous cleft palate is challenging to diagnose owing to the imperceptible presence of this deformity. The present case report shares this challenging diagnosis and management. This case study is interesting to note how perceptual assessment of speech may help in diagnosis and management. The aim of the study is to explore the theoretical bases of submucous cleft palate and its associated speech defects. The objective of the study is to determine the association of speech and language impairment in submucous cleft palate in global perspective. A comprehensive management of communication disorder in submucous cleft palate has been given. KEYWORDS: Occult, Submucous, cleft, communication. INTRODUCTION Submucous cleft palate, the cleft of the muscles and/or bone for the roof of the mouth is covered by a mucous membrane, making it difficult to see the presence of the cleft when looking into the mouth. 7 Some children shows signs of a submucous cleft palate (e.g., bifid uvula, midline groove of the hard palate, or nasal sounding speech), while others may show no signs. Therefore, the treatment of submucous cleft palate is quite variable. 6,11 111

2 A submucous cleft is caused by several factors. In most cases, there is a combination of genetics (inheritance) and environmental factors during the first few weeks of pregnancy. 8 Submucous cleft can occur as part of a genetic syndrome that causes other congenital anomalies. The most common syndromes associated with submucous cleft are Stickler s syndrome and velocardiofacial syndrome (also called 22q.11 deletion syndrome). 15 A submucous cleft palate is a relatively rare variant of the congenital malformation cleft palate The estimated incidence of submucous cleft is 1 in 1200 to 2000 live births. 13 According to two studies done on the populations of normal primary school children for submucous cleft palate, the incidences were- 1:1200 (0.08%) 20 and 1:6000 (0.02%). 4,20 CLEFT PALATE AND ITS EFFECTS- Children with submucous cleft palate face a variety of challenges, depending on the type and severity of the cleft. 18 One of the most immediate concerns after birth is feeding. While most babies with cleft lip can breast-feed, a cleft palate may make sucking difficult. In some cases, a submucous cleft can cause difficulty with sucking. As a result, infants may take a longer time to eat. They may also have fluid come through the nose occasionally. The biggest concern with submucous cleft is its possible effect on speech.5 Most individuals with a submucous cleft have normal speech. However, a submucous cleft can cause velopharyngeal insufficiency (VPI). VPI is a condition where the defect in the velum (soft palate) 3,10 prevents it from closing against the pharyngeal wall (back wall of the throat) while talking, this can cause hypernasality or nasal air emission during speech. 12,13,21 CLASSIFICATION Submucous cleft is mainly divided into two categories which include the classic submucous cleft palate and the occult submucous cleft palate. The classic submucous cleft palate have triad of overt physical finding which includes bifid uvula and furrow along midline of soft palate with attenuated midline raphe, short palate with midline muscle separation and a notch in posterior margin of hard palate. 4 The functional findings in classic submucous cleft palate are the hypernasal speech and velopharyngeal insufficiency. The other type of submucous cleft is Occult submucous cleft palate. In this type there is muscle malposition in the absence of overt physical findings and hypernasal resonance during speech. 4,14 METHODOLOGY: Brief History A very vibrant typically developing child, she had occasional nasal regurgitation which was often ignored and believed to be accidental. Her mother often felt that her child s voice is different but never seek any help for the same. Until and unless the hypernasality became more prominent as her vocabulary increased. She was taken to general physician where they found no other complication except adenoids. Accidently one day an otolaryngologist heard her voice and suggested further radiological assessment. Finally at 2yrs and 2months of age she was diagnosed to have submucous cleft palate which extending from hard palate upto uvula. The surgical treatment was done at the age of 2 year 3 months of age. Palatoplasty surgery was done by Veau-Wardil-Kilner (V-W-K) technique with Intravellar Veloplasty. 7 Post surgery the child was referred for speech and language therapy. Speech and Language evaluation The assessment started with a detailed case history session which showed no significant pre-natal and peri-natal history, and was identified to have submucous cleft at 2 years 2 month of age. The motor and speech and language development was age appropriate as reported. The oral peripheral mechanism examination showed lips to be normal in appearance and function (puckering, retraction, and seal). The appearance and functioning of tongue was normal (Protrusion, Lateral movement, Elevation, Retroflexion was adequately present) teeth and mandible was normal in appearance and function. Hard palate examination revealed repaired cleft; length of the palate was short, width was normal, and ability to degluttate was adequate. Soft palate showed repaired cleft, movement during the production of /ah/ was sluggish, ability to suck through straw was inadequate, ability to blow paper bits/candle balloon/soap bubbles was feebly present. Gag Reflex was present and uvula was short in length. Hypernasality was present. Facial symmetry was present. Modified Striped Y Elsahy classification showed region 9, 10 and 11 i.e., Hard palate posterior to incisive foramen and Soft palate repaired by surgery. Language evaluation revealed age appropriate language skills. Speech and language therapy The Speech and language programme was based upon the results of the assessments done. The baseline was compensatory productions which included poor oral resonance with hypernasal voice quality. PRAAT showed formation of antiformants in oral sound indicative of hypernasality. Nasal flutter test revealed hypernasal voice. Universal parameters ratings for reporting speech 112

3 outcomes in cleft palate showed a rating of 3 indicative of severe hypernasality. Nasal emission s mirror test showed presence of nasal emission. Universal parameters ratings for reporting speech outcomes in cleft palate showed a rating of 3 indicative of severe nasal emission. Misarticulation Bzoch error pattern articulation test showed; nasalized plosives, nasalized vowels, pharyngeal plosives substituted for the velar plosives k/g, palatal-dorsal productions (mid-dorsum palatal stops), /p/ (bilabial, unvoiced, stop-plosive) substituted with /m/ (bilabial, voiced, stop-plosive, nasal) and /d/ (labiodental,voiced, plosive ) substituted with /t/. Duration of session was 45 minutes, once in a week. Language used for stimulation was Hindi. The Long Term Goal 6 was to facilitate 50% reduction of glottal stops and 50% correct production of stop consonants and to facilitate age appropriate resonance after 12 sessions of therapy and the Duration of plan was 3 months (12 sessions). The short term goals were to reduce hypernasality, nasal air emission. It further included reduction of misarticulation by 50% reduction of glottal stop, facilitation of production of phonemes /d/ (labiodental, voiced, stop-plosive) and /p/ (bilabial, unvoiced, stopplosive), facilitation of production of velar plosive /k/ and /g/ and to facilitate correct production of /ʃ/ and to facilitate increase in the mean length of utterance. The principles which were followed during therapy 8,16,17 was not to use blowing exercises, sucking exercises, velar exercises or oromotor exercises since the problem is rarely muscle weakness and these exercises do not work. Pinching of nose to try to improve velopharyngeal function was avoided because closing the nose actually makes it impossible for the velum to go up. The figure 1 below mentioned target sounds according to hierarchy was chosen. Figure 1 : HIERARCHY OF TARGET SOUND SEQUENCE HIERARCHY OF TARGET SOUND SEQUENCE 6 Break glottal pattern with h front (lip) low pressure, nasal (/w/, /m/) (/p/, /b/),(/f/, /v/) -alveolar ) low pressure (/y/,/n/) -alveolar ) high pressure (/t/,/d/) -interdental ) /ɵ/ -alveolar ) /s/,/z/ -tip-palatal) high pressure /ʃ /, / ʈʃ /, / j/ ( tongue tip retroflex ) /r/ The first target behavior was hypernasality and short term goal was to encourage oral resonance in order to reduce hypernasality. Yawning and open mouth approach 19 was used. During the activity a listening tube was used with which the child was made to discriminate between oral and nasal sounds. The child was made to put one end of the tube in the entrance of the nostril and the other end near her ear. When nasality occurred it was very loud through the tube. The child was asked to reduce or eliminate sound coming through the tube as she produced oral sounds and words. Similarly one end of the listening tube was put in front of the child s mouth and the other end near the child s ear. The child was asked to try to increase the oral pressure on the oral sounds and hear it through the tube. The second activity was yawning, followed by a vowel /i /-target consonant (flattens base of tongue and elevates soft palate). 113

The second target behaviour was Nasalized plosives, nasalized vowels. The short term goal was to encourage oral resonance. Yawning, cul-de- sac techniques were used.

4 The second target behaviour was Nasalized plosives, nasalized vowels. The short term goal was to encourage oral resonance. Yawning, cul-de- sac techniques were used. The child was asked to produce a big yawn, which pushes the back of the tongue down and the velum up. The child was made aware of the stretch in the back of his mouth. During the activity the child was made to articulate the nasalized sound (vowel, bilabial plosive or lingual-alveolar plosive, or /l/) with the yawn, while feeling the stretch in the back of the mouth. Auditory feedback was given at the same time using listening tube or the Oral nasal listener. The third target behavior was nasal emission and the short term goal was to reduce nasal emission. Visual feedback and cul-de-sac technique was used. Handmade SEE-SCAPE, shown in Figure 2 was self developed. It is a device with a vertical tube and a light weight ball in it that rises when air is blown into it through the nose from another flexible tube that the child put near nose. The device is used to make the child realize about the air escaped through her nose while speaking and she was encouraged to reduce or stop the movement of the ball in the tube by delivering the air towards the oral cavity. Figure 2: Indigenous See-Scape The fourth target behavior was Glottal-stops production.the short term goal was 50% reduction of glottal stop; the child was made to produce the voiceless plosive and then the vowel preceded by a /h/. For example, p...ha for pa and p...ho for po. This keeps the vocal folds open and prevent the glottal stop. Gradually the transition time decreased from the consonant to the vowel until the syllable is produced without glottal stop. This activity was practiced from the first day of the therapy. The fifth target behavior was substitution of /p/ and /d/ phonemes with /m/ and /t/ and the short term goal was to facilitate production of phonemes - /p/ (Bilabial, unvoiced, stop-plosive) and /d/ (labiodental, voiced, plosive). Phonetic placement method 1 and Minimal pair approach 1 was used for /p/. The sound was modeled several times. Attention was drawn towards the lip closure, building up pressure in the mouth, and air explosion as the sound is produced. The lips were manually guided to the required articulatory posture. Minimal pair approach was used to differentiate between /p/ and/m/. 1 This activity was practiced from the first therapy session. For /d/, the child was made to practise words with the phoneme /d/ like- (doll, didi, daal) and was made to add voicing while saying /t/. This activity was practiced from the 3rd therapy session. The sixth target behavior was Pharyngeal plosives substituted for the velar plosives k/g and the short term goal was To Facilitate production of velar plosive /k/ and /g/ Phonetic placement (Establishment of placement for velar plosives (/k/ and /g/) by starting with an /ng/ a spoon upside down was used to hold the tip of the tongue down. Then the chin was firmly pressed with thumb to push the tongue up. Once /ng/ placement was established, the child was then made to drop the tongue. Work on the up and down movement of the back of the tongue was done to replace the back and forth movement which occurs with the pharyngeal plosive. Once the child produced the /k/, voicing was added for /g/.this activity was practiced from the 5th therapy session. The seventh target behavior was Palatal-Dorsal Productions (Mid-Dorsum Palatal Stops). The short term goal was correct production of /s/. A straw was placed at the front of the child s closed incisors and was asked to produce a /s/ and was asked to listen to the air stream that goes through the straw. The straw was moved to the side of the child s dental arch during production of the /s/, and the child was asked find the place where the air stream can be heard through the straw. The child was asked to put the straw at the front of his closed incisors and produce a /t/ while keeping the teeth closed. The child was asked to push the air into the straw at the front of 114

5 the teeth and hear the air through the straw. After achieving this position, the child was asked to prolong /s/ without using /t/. This activity was practiced from the 8th therapy session. The eighth short term goal was to facilitate increase in mean length of utterance. Enhanced Milieu Teaching was used. The child was motivated to practice language when there is functional consequence. Verb cards and toys were used. Opportunities for functional language were created by arranging the environment to facilitate requests. Prompts were given and new words were modeled in response to requests. To encourage practice of new words, adequate expansion, praise and access to requested objects were provided. Phonological recasting was done for correcting child s utterances. Environmental arrangement was provided like a naturalistic environmental setting was created such as an object (toy or stickers) which the child likes were placed at a place where she cannot reach but can see. Mand-model like prompt for a communicative response was given. Real questions like what do you want?, what will you do with it? were introduced. An instruction to verbalize preference or give information was given like do you want a toy? `. An opportunity to indicate a choice was provided like do you want an eraser or sharpener? Time delay was monitored such as a time delay of 5 seconds was given to the child for giving response and was also reinforced to initiate responses. STATUS REPORT AFTER 12 SESSIONS Universal Parameters Ratings for Reporting Speech Outcomes in Cleft Palate 8 The parameters showed slight decrease in Hypernasality from level 3 (severe) to level 2 (moderate) in both single words and sentences. Decreased nasal air emission from frequent nasal air emission to intermittent in both single words and sentences is also noticed. There was decrease in consonant production errors. Increase in overall intelligibility from level 3 (severe) to level 2 (moderate) in both conversational speech and whole speech sample were rated. BZOCH ERROR PATTERN ARTICULATION TEST 2,9 Increased intelligibility of plosives and glides which were indistinct due to nasal emission. Decrease in simple and gross substitutions mainly in the initial and in the final position was seen. Table 1: Pre and post therapy articulation data after 12 sessions PRE-THERAPY POST-THERAPY Nasal emission test 10/10 Nasal emission test 7/10 Hypernasality test 10/10 Hypernasality test 7/10 Hyponasality Test 0/10 Hyponasality Test 0/10 Phonation test /i/ 6 sec Phonation test /i/ 5 sec 115

CONCLUSION Submucous cleft palate often gets unnoticed because of the lack of visibility of this deficit. Hence the intervention gets delayed and prognosis is hindered.

ACKNOWLEDGEMENT We are thankful to the parents of this child for their generous contribution and dedication. REFERENCES 1. Barlow, J. A., and Gierut, J. A. (2002).

6 CONCLUSION Submucous cleft palate often gets unnoticed because of the lack of visibility of this deficit. Hence the intervention gets delayed and prognosis is hindered. In such situation presence of cleft speech may help in diagnosis and intervention early. The present case study is an example of the same. ACKNOWLEDGEMENT We are thankful to the parents of this child for their generous contribution and dedication. REFERENCES 1. Barlow, J. A., and Gierut, J. A. (2002). Minimal pair approaches to phonological remediation. Seminars in Speech and Language, 23(1), Bzoch KR (Ed.): Communicative Disorders Related to Cleft Lip and Palate, 5th Ed., Pro-Ed, Austin, D Antonio LL, Eichenberg BJ, Zimmerman GJ, Patel S, Riski JE, Herber SC, Hardesty RA. (2006). Radiographic and aerodynamic measures of velopharyngeal anatomy and function. Plast Reconstr Surg., 106(3): Garcia Velasco M, Ysunza A, Hernandez X, Marquez C. Diagnosis and treatment of submucous cleft palate: a review of 108 cases. Cleft Palate J. 1988; 25: Golding-Kushner KJ, Argamaso RV, Cotton RT, Grames LM, Henningsson G, Jones DL, et al Standardization for the reporting of nasopharyngoscopy and multiview videofluoroscopy: A report from an International Working Group. Cleft Palate Journal 27(4): Golding-Kushner, K.(2001). Therapy Techniques for Cleft palate Speech & Related Disorders, Singular Thomas Learning, San Diego. 116

7 7. Gosain AK, Conley SF, Marks S, Larson DL (1997). Submucous cleft palate: diagnostic methods and outcomes of surgical treatment. Plast Reconstr Surg, Gunilla Henningsson, David P. Kuehn, Debbie Sell, Triona Sweeney, Judith E. Trost -Cardamone, Tara L. Whitehill, (2008). Universal Parameters for Reporting Speech Outcomes in Individuals with Cleft Palate. The Cleft Palate- Craniofacial Journal. 45(1): Harding, A, & Grunwell, P. (1998). Active versus passive cleft-type speech characteristics. Int. J. Language & Communication Disorders, 33(3), Hess, D., (1976). A new experimental approach to assessment of velopharyngeal adequacy: Nasal manometric bleed testing. J. Speech Hear. Dis., 41, Cited in Rampp, D. L., Pannbacker, M., Kinnebrew, M.C., (1984). Velopharyngeal Incompetency: A Practical Guide for Evaluation and Management. Austin: Pro-Ed. 11. Kummer A. (2007). Cleft Palate & Craniofacial Anomalies, 2nd Ed., Delmar Cengage Learning, New York. 12. Kummer, A. W. (2006). Resonance disorders and nasal emission: Evaluation and treatment using "low tech" and "no tech" procedures. The ASHA Leader, 11(2), 4, Kummer, A.W. (2008). Cleft Palate and Craniofacial Anomalies: Effects on Speech and Resonance (2nd ed). NY: Thomson Delmar Learning. Describes compensatory articulation productions 14. McWilliams BJ (1991).Submucous clefts of the palate: how likely are they to be symptomatic? Cleft Palate Craniofac J.;28: Peterson-Falzone, S., Hardin-Jones,M., Karnell, M., Cleft Palate Speech (2001), 3RD Ed., Mosby, St. Louis. 16. Peterson-Falzone, S., Trost-Cardamone, J., Karnell, M., Hardin-Jones, M., (2006 ). The Clinician s Guide to Treating Cleft Palate Speech, Mosby, St. Louis, 17. Peterson-Falzone, S., Trost-Cardamone, J.E., Karnell, M. & Hardin-Jones, M. (2005). The Clinician s Guide to Treating Cleft Palate Speech. NY: Mosby. 18. Roopa Nagarajan, V. H. Savitha, and B. Subramaniyan(2009). Communication disorders in individuals with cleft lip and palate: An overview. Indian J Plast Surg, Oct; 42(Suppl): S137 S143.. doi: / Stemple, J., Glaze, L., Klaben, B.,(2010). Clinical Voice Pathology Theory and Management, 4th Ed., Plural Publishing, San Diego. 20. Weatherley-White RC, Sakura CY Jr, Brenner LD, Stewart JM, Ott JE (1972). Submucous cleft palate: its incidence, natural history, and indications for treatment. Plast Reconstr Surg, 49: Weiss, C., (1974). The speech pathologist s role in dealing with obturator-wearing school children. J. Speech Hear. Dis., Cited in Rampp, D. L., Pannbacker, M., Kinnebrew, M.C., (1984). Velopharyngeal Incompetency: A Practical Guide for Evaluation and Management. Austin, TW: Pro-Ed. 117

Cleft Palate Speech-Components and Assessment Voice and Resonance Disorders-ASLS-563. Key Components of Cleft Palate Speech.

Cleft Palate Speech-Components and Assessment Voice and Resonance Disorders-ASLS-563 Key Components of Cleft Palate Speech Disorder Type of Disorder/ Causes Hypernasality Resonance Disorder insufficiency