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CASE REPORT
An unusual etiology of velopharyngeal insufficiency M-L Haapanen’, L Ertama2, AE Pesonen’, M Paaso’ and H Hagman*
’Cleft Centre, Department of Plastic Surgery, Helsinki University Central Hospital, and The Department of Radiology, ENT hospital, Helsinki University Central Hospital.
Scand J Log Phon 20: 151-155, 1995.
Velopharyngeal insufficiency is usually associated with cleft palate. However, there are also other causes for signs of velopharyngeal insufficiency in speech. We describe an unusual etiology of hypernasality and nasal air emissions in a 5-year- old boy. Based on clinical, logopedic, neuropsychological, nasometric and videofluoroscopic examinations we concluded that velopharyngeal insufficiency in the present patient was associated with clutter-stuttering. We emphasize the need for thorough examinations of the etiology of velopharyngeal insufficiency in children without cleft palate for designing adequate treatment. Key words: velopharyngeal insufficiency, hypernasality, stuttering, cluttering.
Marja-Leena Haapanen. Cleft Center, Department of Plastic Surgery, Helsinki University Hospital, Topeliuksenkatu 5, 00260 Helsinki, Finland.
INTRODUCTION
The most common etiology of velopharyngeal insuffi- ciency (VPI) is cleft palate or conditions after palatal repair. Signs of VPI in speech may sometimes be due to a congenitally short palate, or a deep or enlarged pharynx. Hypernasality may also follow adenotonsil- lectomy (Donati et al., 1991, Gibb, 1969, Michel and Brusis, 1990). Neuromotor deficits of the palate and pharynx may be responsible for severe and difficult-to- manage cleft-like speech (McWilliams et al., 1990). In the present study we describe a 5-year-old boy with an unusual etiology of hypernasal speech.
THE SUBJECT
A boy of 5 years and 10 months of age was referred to Cleft Center because of hypernasality and audible nasal air emissions not associated with cleft palate. The child had received 16 sessions of speech therapy at his kindergarten, but hypernasality or audible nasal air emissions did not cease.
History: A physically normal child was born 4 weeks before the date by section. According to the mother, the boy started to utter his first words at age 12 months, and sentences at age 1.5 years, but the speech remained unclear. The child started to walk at the age of 1.3 years. According to the mother the speech of the child became disfluent when he started kindergarten at the age of three years.
Disfluency occurred when the child was uncertain of the attention or approval of the adults with whom he was speaking. If the child felt secure and relaxed, there
was no disfluency according to the mother. The mother felt that speech therapy impaired the dis- fluency. The mother had herself an unclear speech until school age. The child was left handed.
THE EXAMINATIONS
At the Cleft Centre the following examinations were performed in addition to interviewing the mother. Clinical examination which included evaluation of speech and the anatomic structure of articulatory organs, was performed by the speech pathologist (the first author) and the speech therapist (the fourth author) of Cleft Center. Neuro-psychological tests were performed by a psychologist (the third author). The following instrumental examinations were done: nasalance measurements (Fletcher et al., 1989, Haapanen, 1991a) by the Nasometer Model 6200-2, and videofluorographic examinations of velo- pharyngeal functioning.
Speech material The assessment of the quality of speech was based on the analysis of continuous spontaneous speech, a Finnish articulation test (Remes, 1975), and 8 test sentences ffiivi heijaa vauvaal (Viivi swings a baby), /Olli laulaa Lailaa/ (Olli sings a song called Laila), /Atte otta tuttis/ (Atte takes a pacifier), Pappa puhuu puppua/ (Daddy speaks nonsense), /Assi sossottaa/ (Assi speaks poorly), Paula harjasi tukkaa/ (Paula combed her hair), /Kukko kiekuu Kukko kiekuu/ (A cock cries doodledoo), /Kuka tekee papupataa/ (Who
01995 Scandinavian University Press. ISSN 0803-5032 Scand J Log Phon 20
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makes bean soup), /Minne mummo meni/ (Where did grandma go?). The same test sentences were used for the perceptual speech analysis, the nasalance measure- ments and for the videofluorographic analysis.
RESULTS
Fluency of speech At the first examination at the Cleft Centre the child spoke somewhat anxiously. He spoke much and quickly, as if he had tried to utter as many words as possible by one expiratory phase. Sometimes the sentences or parts of them were inhibited. The child experienced various nonspeech behaviours and move- ments described by Conture and Kelly (1991), such as audible outbreathing, swinging of his legs or bending of his head forward as if to help the words come out of his mouth. The child appeared to have remarkable difficulties in repeating some of the test sentences, such as /Olli laulaa Lailaa/. It seemed to be very difficult for him to utter a word which begun with an /l/ sound, although he could pronounce the ill sound properly in isolation. He experienced both clonic repetitions and tonic inhibitions of most of the speech utterances including /l/. The repetitions were very rare, but the inhibitions were more common. The repetitions and inhibitions occurred particularly at the stress peak of the utterance in sentences including an /1/ sound. In other type of sentences the disfluency was very uncommon, and, if present, very mild, being mostly a hardly detectable lengthening of the time interval between the words.
The disfluency was, however, situation dependent.
Figure 1. Lateral videofluorographic view of the velopharyngeal mechanism in a 5-year old boy uttering the sentence /Kuka tekee papupataa/. The view shows the articulators during the first segment of the first word. No stuttering is noted and adequate velopharyngeal closure is visualized.
It occurred during the first speech assessment sessions, and ceased later. Finally the child experienced only mild phonemic specific tonic inhibition of utterances.
The boy also had difficulties in repeating test words reflecting verbal practice skills, such as /pata/ or /pata ka/. He was able to repeat only twice the words /pata/ and once /pataka/.
Phonological errors: The child actually had no other phonological errors than evolutional errors of vowels /ae, oe, y/. He uttered those front vowels as their back counterparts. The liquid /r/ was a slightly inaccurate anterior variant of the standard phoneme. Syntagmatic phonological errors, such as described by (Nettelbladt, 1983), were not found.
Signs of velopharyngeal insufficiency: Clear and severe signs of VPI were found in clinical speech examination. The signs were not, however, generally present during production of isolated words or test sentences. Signs of VPI-occasional hypernasality and audible nasal air emissionsnccurred while the child spoke spontaneously, and they were worst at the first examination session.
Nasalunce measurements: Nasalance scores were higher than normal in the first examination session, but not during further examinations. At the first examina- tion the mean nasalance score for the sentences with no nasal phonemes was 39 referring to moderatehevere nasality and in further examinations the mean score for the nonnasal sentences was 13 referring to normal nasality (Haapanen, 1991a, b). The child had difficul- ties in uttering a sentence loaded with /1/ sounds (/Olli laulaa Lailaa/). The words were inhibited, and he had to
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move and swing his head to help producing the words. At the first trial he scored 47 for that sentence.
Structure The anatomical structure of the articulatory organs was normal on the basis of peroral visual inspection. The soft palate was long with good motility. No palpable defect in the hard palate, and no signs of submucous cleft palate were found.
Videofzuorographic examination: During relaxed production of test sentences in lateral view, good velar movements were observed with closure against the posterior pharyngeal wall (Fig. 1). In frontal view, good shelf-like, medial adduction of the lateral walls were seen. However, two sentences were difficult to produce: /Olli laulaa Lailaa/ and /Paula harjasi tukkaa/. The videofluorographic views showed that articulators started to move towards correct positions for speaking. However, the child could not say a word. He bended his head and changed his posture, before he was able to start speaking, however, without control of the articular movements. He also lost the control of the velo- pharyngeal valving. The velopharyngeal sphincter remained open. It is possible that the /1/ sound provoked the disfluency of his speech. Fig 2 shows the lateral projection of articulatory organs while trying to utter the word /Lailaa/ in the sentence /Olli laulaa Lailaa/.
Neuropsychological investigations: The child under- went neuropsychological investigations, which included the WPPSI test, parts of a Finnish neuropsy- chological test battery (Korkman, 1988), the VMI test (Beery and Buktenica, 1967), and the Token test (DiSimoni, 1978). His nonverbal performance was on
Figure 2. Lateral videofluorographic view in a 5-year old boy uttering the sentence /Olli laulaa Lailaa/. The view shows the inhibition of the velopharyngeal closure due to stuttering during the first sound of the word /Lailaa/.
the average normal. The verbal test profile was uneven. Performance on the vocabulary and information items was good, but logical verbal reasoning and the concept of numbers were not age appropriate. He had slight difficulties in similarities. The visual reasoning was normal, and the spatial constructional ability was good. He also managed well in accuracy of visual percep- tions. Auditory analysis'of speech and comprehension of verbal instructions were normal. In the Token Test he did not achieve age level in comprehension of concepts and naming colours. Digit span was narrow, but logical learning was good. Visual spatial function- ing was good. He had difficulties in kinesthetic praxis and in finger differentiation and visuomotor functions. In addition to various neuropsychological findings the boy had obvious attention deficit disorder (ADD). The child also seemed to have impaired selfconfidence reflected by the fact that he spoiled the drawings he had made. To conclude, the boy had slight pathological findings, mainly in the motor development, and attention deficit disorder.
DISCUSSION
Velopharyngeal insufficiency has been hypothesized to be associated with various risk factors such as hearing impairment, intelligence deficiency, neuro- motor dysfunction and cognitive defects. As far as the present authors are aware, there are no reports on hypernasality associated with disfluency of speech.
Cluttering-stuttering (tachyphemic-dy sphemic speech disorder) has been described as an entity of a congenital language disability syndrome by Lus-
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chinger and Arnold (1970). This speech disorder beings with delayed language development, and passes through stages of severe dyslalia, dysgrammatism or dyslexia, and finally results in cluttering. The congenital cluttering may, on the basis of inborn language disability, change into the secondary reaction of stuttering under the additional effect of unfavorable environment or as a result of faulty attempt at correction. Slight neuromotor dysfunctions are asso- ciated with this disfluency disorder of speech. Dyspractic articulatory behaviour is common in children whose language development is considered congenitally disabled. Inhibition of verbal expression may be aggravated by environmental stress. In this case there is also some overlap with developmental dyspraxia. Also according to the current research (for reviews, see Johannsen et al., 1994, Grnroos, 1995) stuttering is considered to result from combination of various factors such as poorness of neuropsysiological and neuroanatomic development, delayed cerebral dominance of speech, psychosocial or neuropsycho- linguistic problems.
Based on the results of clinical speech evaluation, videofluorographic examination of velopharyngeal valving mechanism, and neuropsychological test performance, a diagnosis of a tachyphemic-dysphemic speech disorder was given to the present patient. Short digit span, finger agnosia, impaired kinesthetic praxis, impaired logical verbal reasoning and the left handedness were considered to reflect slightly imma- ture or deviant development of central nervous system. Signs of velopharyngeal insufficiency in speech was attributed to the phases of cluttering-stuttering. Cluttering-stuttering occurred with non-speech be- haviours typical in stutterers (Conture and Kelly, 1991). Stuttering may occur in connection with linguistic prerequisites such as syllabic stress (Prins et al., 1991). This was true also in the present patient. The mild delay of the child’s neuropsycholinguistic development was considered to result in learning disabilities at school age. The child was considered to need supportive special teaching at school.
The child was concluded to have primarily normal velopharyngeal structure. A small central opening in velopharyngeal isthmus during speech such as occult submucous cleft palate might have theoretically induced audible nasal air emissions during speech. Nasendoscopic examination would have falsified or verified this hypothesis, but the child was too young to co-operate adequately in that examination. However, the present examinations gave strong evidence for the present hypothesis, that the child’s VPI was associated with disfluency. No need for surgical treatment was found. In this stage we did not recommend speech
therapy, but neuropsychological rehabilitation for the child and psychological counceling for the parents.
CONCLUSION AND CLINICAL IMPLICATIONS
Briefly, the present child was a clutterer-stutterer, who temporarily demonstrated signs of VPI associated with disfluency of speech. Speech disorder was due to slight cognitive and neuromotor disorder. He had an attention deficit disorder probably provoked by the neuropsychological defects and the anxious feedback from the environment.
This case report indicates that dysfluency may result in signs of VPI. Dysfluency, on the other hand, may be associated with neuromotor dysfunctions (Johanssen et al., 1994, Grnroos (1995, a review). Different reasons for VPI should be carefully identified to avoid unnecessary surgery. Uncommon reasons for VPI should be reported to help other clinicians to solve the problems of VPI in cases with no cleft palate.
REFERENCES
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Beer K, Buktenica NA. Developmental test of visualiza- tion intergration. Cleveland: Modern curriculum press. 1967. Conture EG, Kelly EM. Young stutterers’ nonspeech behaviours during stuttering. JSHR 1991; 34:
DiSimoni F. The Token test for children-Manual Teaching Resources Corporation, Massachusetts. 1978. Donati F, Pfammatter JP, Mauderli M, Vassella F. Neurologische Komplikationen nach Tonsillektomie. Schwez. med. Wschr. 1991; 121: 1612-1617. Fletcher SG, Adams LE, McCutcheon MJ. Cleft Palate speech assessment through oral-nasal acoustic measures. In KR Bzoch (Ed.). Communicative disorders related to cleft lip and palate, pp. 246-257. 3rd ed. Boston: College Hill Publication, Little Brown and Company. 1989. Gibb AG. Unusual complications of tonsil and adenoid removal. J Laryngol Otol 83 1969; (2): 1159-1174. Gronroos M. Ankytys. Duodecim 1995; 11 1: 560-565. Haapanen M-L. A simple clinical method of evaluating perceived hypernasality. Folia Phoniatr 1991a; 43:
Haapanen M-L. Nasalance scores in normal Finnish speech. Folia Phoniatr 1991b; 43: 197-203. Johannsen HS, Schulze H, Rommel D, HZgel A. Stuttering in childhood: A five-year longitudinal study in progress. Folia Phoniatr Logop 1994; 46: 241-249. Korkman M. NEPSU-Lasten neuropsykologinen tutki- mus. Uudistettu versio. Kasikirja. Helsinki: Marit Korkman ja Psykologien kustannus Oy. 1988. Luchsinger R, Arnold E. Voice-Speech-Language, B special part, chapter V pp. 739-768. Belmont, California and London: Wadsworth Publishing Company. 1970. McWilliams BJ, Morris HL, Shelton RL. Cleft Palate meech. Philadebhia: BC Decker Inc. 1990.
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15. Nettelbladt, U. Developmental studies of dysphonology in children. Travaux de L'institute de linguistique de Lund XIX. Lund: Gleerup. 1983.
16. Prins D, Hubbard CP, Krause M. Syllabic stress and the occurrence of stuttering. JSHR 1991; 34: 1011-1016.
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SAMMANFATTNING
En ovanlig orsuk till velofuryngeal insufjiciens: Full- beskrivning Velopharyngeal insufficiens (VPI) hanger i allmanhet samman med gomspalt, men andra orsaker ar valk- anda. Vi beskriver en ovanlig etiologi till oppen nasalering och genomslag hos en femdrig pojke. Efter en utredning innefattande kliniska, logopediska, neuro- psykologiska, nasometriska och videorontgenologiska undersokningar kom vi fram till att patientens VPI
mdste hanga samman med hans stamning och skenande tal. Vi understryker behovet av ingiende utredningar av barn med VPI utan gomspalt som utgingspunkt for beslut angdende behandlingen.
YHTEENVETO
Eputavallinen honotuksen syy Suulaen ja nieIun yhteistoiminnan vajaus liittyy yleensa suulakihalkioon tai sen jalkitilaan. Kuvaamme tutkimuksessamme 5-vuotiaan pojan, jolla puheessa honotuksena ja nenaturinoina ilmeneva velofaryngaa- linen insuffisienssi aiheutui poikkeuksellisesta syysta. Kliinisen, akustisen, radiologisen, foniatris-logo- pedisen seka neuropsykologisen tutkimuksen perus- teella voitiin todeta, etta potilaamme velofaryngaali- nen vajaatoiminta liittyi puheen sujuvuushairioon, joka puolestaan liittyi ankytys-sokellukseen. Tapaustutkimuksemme osoittaa, etta honotuksen syy on huolellisesti selvitettava virheellisten hoitoratkai- sujen valttamiseksi.
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