British Journal of’Plasric Surgery ( 1990), 43, 187- 196 8 1990 The Trustees of British Association of Plastic Surgeons
The role of pharyngoplasty speech disorders P. M. DAVISON,
in congenital
neurogenic
R. E. RAZZELL and A. C. H. WATSON
Royal Hospital for Sick Children, Edinburgh Summary-Children with dysarthria are often seriously handicapped by their inability to communicate. Velopharyngeal incompetence may contribute to their speech difficulties but the indications for pharyngoplasty are poorly defined. The speech of 41 children, thought to have a defect of neurological origin and referred for possible pharyngoplasty, was assessed by a speech therapist, by nasopharyngoscopy and by multiview videofluoroscopy. These investigations revealed that 16 had velopharyngeal incompetence, with a neurological aetiology, and might be expected to benefit from pharyngoplasty. Independent assessment of the intelligibility of these 16 children before and after pharyngoplasty has been related to their preoperative oropharyngeal function. The majority benefited from surgery, some substantially. In particular, good lip and tongue function are shown to be necessary to achieve improved intelligibility. The roles of age, intelligence and aetiology in the success of pharyngoplasty are discussed.
It was shown by Randall et al. (1960) that pharyngoplasty can improve the speech of patients with velopharyngeal incompetence due to neurological disorder. However, few guidelines on the indications for surgery have since emerged. It has been suggested by Heller et al. (1974) that reducing nasal emission and improving nasality will not necessarily lead to better intelligibility, unless dysarthric speech patterns can be amended. To date, most authors when discussing the surgical management ofdysarthric speech have documented changes in voice quality rather than changes in speech intelligibility as recommended by Johns (1982). Few papers have attempted to relate these changes to the function of structures involved in articulation. The dual objectives of this study were (1) to investigate, by assessment of their intelligibility, whether pharyngoplasty can benefit the speech of dysarthric children; (2) by quantifying function of the pharynx, the velum, the tongue and the lips, to identify any factors which could lead to better patient selection.
investigation of speech problems neurogenic origin.
of suspected
Initial assessment
A thorough speech assessment, as detailed in Table 1, was carried out on each child by an experienced speech therapist (R.E.R.). Video recordings were made of conversational speech, and nasopharyngoscopy and multiview videofluoroscopy performed. The function of each of the structures involved in articulation, i.e. the lips, the tongue and the soft palate together with that of the lateral pharyngeal walls and the posterior pharyngeal wall, was quantified (Fig. 1). Velar, lateral pharyngeal and posterior pharyngeal wall movements were judged by a combination of endoscopy and videofluorosTable 1
Methods of assessment of speech and language
Receptive language Expressive language Articulatory movements Articulation Nasal escape
Material and methods
Nasal resonance
Over a 7 year period, 1979-1986, 41 children were referred to the Edinburgh Cleft Palate Clinic for
Nasal grimacing
187
and snorting
Reynell or Reynell or Imitation EAT Subjective scale Subjective scale Observation
PPVT APT
judgement,
S-pt
judgement,
3-pt
188
BRITISH JOURNAL LATERAL PHARYNGEAL
SOFT PALATE /
\
LIPS
SCORE 0,l
WALL
POSTERIOR PHARYNGEAL
OR 2 FOR EACH OROPHARYNGEAL
OF PLASTIC SURGERY
WALL
MOVEMENT
Fig. 1 Figure l-The Oropharyngeal
summation of the scores Movement Score
from
each of the five functional
copy. Lip function was assessed by asking the children to purse and spread their lips, and tongue mobility by observation of tongue elevation, depression and lateral movement. By awarding 2 for normal function, 1 for reduced rate or amplitude, and 0 for no function for each of these five elements, we arrived at a total that, for the purpose of this paper, we have termed an Oropharyngeal Movement Score with a maximum score of 10. Diagnosis and management ofpatients
Whilst not every child allowed all the investigations to be performed, enough information was gleaned to eliminate 17 patients for reasons shown in Table 2. Twenty-four children were found to have veiopharyngeal incompetence which was contributing to their dysarthria; one was lost to follow-up. The management of the other 23 is shown in Table 3. The 6 children whose velopharyngeal incompetence was found to be intermittent, detectable only during conversational speech but not on isolated sounds, were advised to have further speech therapy. None has since been referred back for reassessment. Another had severe respiratory problems making surgery ill-advised. The ages of the 16 children who had a pharyngoplasty and form the basis of this study ranged from 3 to 15 years (Fig. 2). All had undergone
elements
involved
in articulation
was termed
the
prolonged speech therapy prior to surgery. The causes of their dysarthria, diagnosed by a neurologist, are given in Table 4. The pharyngoplasty was performed on all but one of the children by the same surgeon (A.C.H.W.). The technique used was tailored to the patient’s endoscopic findings. When a large central incompetent port was visible, a posterior pharyngeal hap was used. This was based inferiorly if there was neither palatal nor pharyngeal wall movement, but a superiorly based flap was employed in the presence of large adenoids or if there was some movement of the lateral pharyngeal walls, Patients eliminated from the study as they did not have both velopharyngeal incompetence and a neurological cause Table 2
Palatopharyngeal disproportion Microform cleft palate Immaturity Dysarthric but no velopharyngeal
incompetence
1 2 6 8
The management of dysarthric children who had velopharyngeal incompetence
Table 3
Pharyngoplasty undertaken Unfit for pharyngoplasty Further speech therapy alone
16 1 6
THE ROLE OF PHARYNGOPLASTY
IN CONGENITAL
NEUROGENIC
189
SPEECH DISORDERS
Patient Age when Pharyngoplasty Advocated
Figure 2-The
patient’s
age when pharyngoplasty
was recommended.
in an attempt to place the flap at the level of this activity. A wide transverse port with some palatal and no lateral wall movement was obturated by modified Hynes/Orticochea lateral wall flaps (Table 5). It was recognised that, in the presence of poor movement of both soft palate and pharyngeal walls, it would be impossible to achieve velopharyngeal competence without obstructing the nasal airway, and no attempt was made to do this. The aim was a compromise; to reduce nasal escape to acceptable levels while allowing adequate nasal airflow. This was done by raising flaps of moderate size. After surgery all the children continued with their speech therapy. Postoperative
each patient’s pre- and postoperative speech as recorded on U’matic videotape, with the screen hidden. The items of speech were presented in a Table 4 The causes of dysarthria had velopharyngeal incompetence
in the children
Upper motor new-one Cerebral palsy Arrested hydrocephalus Articulatory dyspraxia Suprabulbar palsy Generalised hypotonia
2 2 2 1 1
Lower motor newone Isolated palatal paresis Moebius syndrome
5 3
who
assesment
Postoperative assessment was a repeat of the complete preoperative work-up, with a follow-up period ranging from 6 months to 3 years, with a mean of 19 months. In addition a comparison was made between pre- and post-treatment intelligibility, as judged by an independent five-person lay panel. The panel members were asked to grade
Table 5 The pharyngoplasty complications
techniques
used, and their
Technique
No.
Mujor complications
Superior pharyngeal Inferior pharyngeal Hynes/Orticochea
6 5 5
Haemorrhage
X 1
190
BRITISH JOURNAL
random order and the panel were asked to place each item on the following 4-point scale : unintelligible poorly intelligible comprehensible normal
= 1 =2 z=3 =4
these specific details were unobtainable preoperatively. Taken together, these results suggest that an effective obturator was constructed in most of the
For each patient the mean intelligibility score was calculated both pre- and postoperatively and the difference between these figures has been used to define the Improvement in Intelligibility as shown in Figures 5, 7 and 8. The significance of these improvements has been tested using the Spearman Rank Correlation analysis and is quoted in the text.
POST
F
Results Initial speech assessment showed that most of the children had delayed receptive and more severely delayed expressive language abilities. Their imitation of articulatory movements and their articulation patterns were poor. All bar one had audibly detectable airflow problems with abnormal resonance patterns; the exception had gross velopharyngeal incompetence visible endoscopically during speech, despite his nasal escape being inaudible. Nasal grimacing and snorting were not outstanding features of the group. A breakdown of each individual child’s results is shown in Table 6. It is interesting to note that most of the children with Moebius syndrome not only had 6th and 7th cranial nerve palsies as is generally recognised, but also had problems with even simple tongue movements. Three out of 5 children referred with an apparently isolated palatal paresis were also found to have significant restriction of tongue and lip mobility. Figure 3 shows a comparison of pre- and postoperative nasal escape (after the method of Boorman and Sommerlad, 1987) measured on a 5point scale by the speech therapist, for fourteen children. Figures in the shaded area would represent patients whose escape had deteriorated with surgery, those in the stippled area the 3 patients in whom no change was detected. The 11 patients in the upper right-hand corner had all improved (p >O.OOl), some substantially. Figure 4 compares nasal resonance before and after surgery. The improvement in both nasal escape and resonance in the 2 remaining children cannot be illustrated as
OF PLASTIC SURGERY
OPERATIVE
SEVERE
2 ii 0
MODERATE
I MILD
NONE
Fig. 3 Figure &There was a significant nasal escape after pharyngoplasty.
POST
(p=O.OOl)
improvement
in
OPERATIVE
HYPER
P
NORMAL
0
E
HYPO
Fig. 4 Figure &Nasal resonance was reduced by pharyngoplasty in some patients. (N.B. Only the numbers in the unshaded vertical represent the intended surgical result).
4f
6
3
Superior
Unintelligible+
8
Superior
5
7
H/O*
Palatal
Unintelligible Poor + normal
2
Superior
6
Palatal paresis
14
Palatal paresis
Comprehensible
6
Superior
I4
Unintelligible Comprehensible
I
Unintelligible
7
Palatal paresis
* Hynes/Orticochea
+ comprehensible
Comprehensible
3
Superior
poor
+ unintelligible
-+ normal
+ normal
+ unintelligible
+ comprehensible
No audible nasal escape, but gross velopharyngeal insufficiency seen on endoscopy
15
-+ comprehensible
Comprehensible
2
H/O*
Inferior
14
II
Moebius syndrome
Palatal paresis
paresis
10
Poor + comprehensible
6
Superior
30
10
6
17
8
24
33
18
36
28
poor
Unintelligible+
+ unintelligible
7
Unintelligible
4
24
6
I
Inferior
by reactionary
22
Length o~followup (mthl
Inferior
+ normal
Flap disrupted haemorrhage
Comments
6
Comprehensible
intelligibilit)
H/O*
H/O*
5
Generalised
4
4
hypotonia
Articulatory
Moebius syndrome
dyspraxia
Articulatory
Moebius syndrome
palsy
dyspraxia
Suprabulbar
5
8
Arrested hydrocephalus
4
4
H/O*
I1
Arrested hydrocephalus
Poor -+ normal
Unintelligible
3
-+ poor
Poor + comprehensible
4
Inferior
palsy
Inferior
Tongue and lip Pre- and postoperative score
I1
15
Articulator) movement score
Kernicterus
Cerebral
details
Pharvngoplasty technique
patient
Age f yr)
Individual
Diagnosis on referral
Table 6
192
BRITISH JOURNAL OF PLASTIC SURGERY
I
I
z
1
POST ;PERATl”E
a
3
IMPROVEMENT IN INTELLIGIBILITY (4 point scale)
Fig. 5 Figure SImprovement in nasal escape could not be related to improvement in intelligibility after surgery (Improvement is defined in the text).
children but that the narrowing of the nasopharynx was probably overdone in 3 patients. None of these suffered any resultant disabling symptoms. There was no obvious correlation between the improve-
POST w
OPERATIVE Y
m
iii
x 5
2
ii G
z
2 5
5
i
e z
UNINTELLIGIBLE
F POOR is
ment in nasal airflow obtained and the type of pharyngoplasty performed. The panel were found to give a remarkably consistent judgement in the change between each child’s pre- and postoperative speech, allowing a quantitative assessment of improvement in intelligibility to be made. Thus a child who had started with poorly intelligible speech and who achieved normal speech after operation, judged as a mean by the panel, was graded as having made a 2+ improvement in intelligibility. Figure 5 shows that the improvements in intelligibility could not be related to the reductions in nasal airflow (p=n.s.) that have been demonstrated. Intelligibility before and after surgery is shown in Figure 6, indicating that none was worse off for having a pharyngoplasty, even though one child’s flap was disrupted by a reactionary haemorrhage; this was the only major complication in the series. Eleven of the 16 children had improved (p = O.OOl),including 5 who achieved normal or near normal speech. Of the 7 children who started with totally incomprehensible speech, 4 could make themselves understood, even if with difficulty. Three children remained unintelligible. Age did not appear to influence the degree of improvement, but an obvious trend is apparent when improvement is collated with muscle function as measured by the preoperative oropharyngeal movement score (p = 0.01) (Fig. 7). An even more emphatic relationship is revealed if just the tongue and lip movement scores are compared with intelligibility (p < 0.01) despite the apparent crudeness of the assessment (Fig. 8). It is interesting to note that tongue and lip function was virtually unchanged by the operation in any of the children. The number of children with each diagnostic label is too small for any relationship to emerge between aetiology and improvement in intelligibility. It was noted that children with upper motor neurone conditions tended to have better control of their tongue and lips than those with lower motor neurone conditions.
kk!
0 w
Discussion
COMPREHENSIBLE
NORMAL
Fig. 6 Figure &Intelligibility by pharyngoplasty.
was significantly
(p=O.OOl)
improved
Speech is fundamental to the developing child’s ability to communicate and is therefore crucial to his behavioural, social and intellectual performance. Serious communication problems lead to reduced self-confidence and frequently to emotional disturbances. Conversational speech needs cerebration both to
THE ROLE OF PHARYNGOPLASTY
IN CONGENITAL
NEUROGENIC
SPEECH DISORDERS
193
Neurological disorders of speech may therefore be divided into three broad categories : -cerebral disturbance (dysphasia) -disorders of the larynx or its innervation (dysphonia) -motor disorders of the articulatory muscles (dysarthria).
12
0
15
3
OROPHARYNGEAL
6
78
P
MOVEMENT SCORE
Fig. I Figure ‘I-The improvement in postoperative intelligibility was related (p = 0.01) to the preoperative Oropharyngeal Movement Score.
understand the spoken word and to formulate and produce a reply in correct syntax. To vocalise this reply, the respiratory and laryngeal muscles produce a vibrating column of air which is articulated upon by the co-ordinated movement of muscles of the pharynx, the velum, the tongue and the lips.
1
. -
0
1
2
TONGUE AND LIP SCORE
Fig. 8 Figure&The best indicator (p < 0.01) of improved intelligibility was the preoperative assessment of lip and tongue function.
Pharyngoplasty can do nothing to influence dysphasia or dysphonia but has been used in the treatment of dysarthria when failure of velopharyngeal closure seems to be a significant factor in causing poor intelligibility. The term dysarthria encompasses a range of conditions which includes flaccid and spastic paralysis, rigidity and ataxia of the muscles of articulation, depending on the site of the neurological lesion. Articulatory difficulties in the child may be caused by a great number of conditions (Brown, 1985), ranging from localised peripheral nerve lesions to gross central nervous system disturbances resulting in disorders of control of articulation. Many pathological states in children are induced by conditions such as asphyxia and jaundice which may produce diffuse neurological damage leading to a global neurological deficit, resulting in a mixture of disorders such as dysarthria, deafness and mental retardation, each of which could be responsible for speech disorders. Moreover, as the muscles involved with feeding and articulation have developed phylogenetically from the laryngeal sphincter, and the nuclei responsible for the innervation of these muscles are closely approximated around the 4th ventricle, it is not surprising that disorders of respiratory control, phonation and swallowing often accompany dysarthria. Thus speech disturbance in a child may well be the result of a combination of dysphasia, dysphonia and dysarthria. This multifactorial aetiological picture leads to great difficulties in predicting the likely benefit of pharyngoplasty for any given dysarthric child (and makes statistical analysis difficult). Only a few studies have attempted to study the role of pharyngeal surgery in the management of patients with neurogenic dysarthria. The results have been variable. Whilst Broadbent and Swinyard (1959) suggested that patients with palatal paresis might be helped by pharyngoplasty, Randall et al. (1960) showed that a reduction in velopharyngeal insufficiency could improve the speech quality of some dysarthric children. In their study the
194 results in a small number of children with isolated and non-progressive palatal paresis varied from the dramatic to the disappointing. One child with associated neurological disorders of adjacent structures gained little benefit. Low intelligence was felt to be a relative contraindication to surgery. A considerable improvement in the speech of 50% of patients with cerebral palsy was reported by Hardy et al. (1969), but the reasons for failure were difficult to identify; neither intelligence nor the severity of neuromuscular involvement was believed to be a prime factor: “The only common variant in the unsuccessful cases . . . is a lack of intrinsic motivation”. Psychological and social readiness for speech improvement, together with parent co-operation, were factors that Minami et al. (1975) emphasised when reviewing their disappointing results in a group of patients of widely differing ages with palatal paresis. They advocated that patients with neurological disorders of neighbouring structures should be excluded from surgery. Heller et al. (1974), who were more enthusiastic about the place of surgery, also emphasised the contribution of both parental and patient co-operation to success. Though they found the neurological diagnosis was irrelevant, they believed the severity of disability and the presence of associated handicaps, together with limited intelligence, mitigated against improvement. In order that any conclusions be drawn from a study, accurate assessment is vital. The standard preoperative work-up used by this Unit enabled us to rule out suspected velopharyngeal incompetence in 8 dysarthric patients, and to identify 6 children whose incompetence was intermittent. Anatomical defects were recognised in 3 children, 2 of whom might well have been missed without nasopharyngoscopy (Kaplan, 1975). Many of the previous studies in this field predated the common use of this investigation (Pigott et al., 1969), with the result that some patients may have been incorrectly diagnosed. It is important that the cause of dysarthria is identified as accurately as possible by a neurologist, not only to prevent the surgeon operating on a patient with progressive disease but also to document the degree of involvement of other systems, such as the respiratory apparatus, as either factor could be a contraindication to surgery. It is interesting to note that, despite a thorough neurological investigation, our work-up revealed a wide range of variation in the involvement of articulatory
BRITISH JOURNAL OF PLASTIC SURGERY
muscles in children with the same diagnostic label. This fact and the small numbers must inevitably complicate any attempt to relate the aetiology of the dysarthria to the results. The intelligibility of all 16 children in this series who underwent surgery had ceased to improve despite prolonged speech therapy. It is known that these patients are unlikely to improve with time (Minami et al., 1975) but it is important to have maximised all the facets of articulation as far as possible, with speech therapy, before recommending surgery. No single surgical approach was adopted as the authors believe that the technique employed should be dictated by the endoscopic and radiographic findings. Seventy-five per cent. of the children had reduced nasal emission following pharyngoplasty, some considerably. Half of the children remained hypernasally resonant, 3 became hyponasal, with only 4 considered to have normal nasal resonance. The improvement in nasal airflow could not be correlated with the recorded changes in intelligibility. When viewed in isolation, it is not surprising that improvement in any one aspect of voice quality is meaningless as so many other factors may also influence speech in these disabled children. We agree with Johns (1982) that measuring changes in intelligibility, rather than voice quality, is of more value in assessing the benefits of surgery. An unbiased judgement of intelligibility was provided by an independent lay panel who had never met any of the patients concerned. In their opinion 11 of the 16 children had improved, a finding which the authors found encouraging. The question arises, “Could the children who did not benefit have been identified preoperatively?” Measurement of the oropharyngeal movement score shows a not unexpected trend-those patients with greater articulatory muscle function are more likely to benefit. To have adopted a cut-off score of 5 would have eliminated all those whose speech failed to improve but also 2 patients who profited from surgery. Evaluation of tongue and lip function alone revealed a clear division between successful and failed cases. No patient who scored less than 3 for these functions improved, while all those who scored 3 or more showed better intelligibility postoperatively. These assessments were easy to perform and to score. This finding can be explained because the operation is designed to influence the function of velum and pharyngeal walls but has no effect on tongue and lips. In the presence of effective
THE ROLE OF PHARYNGOPLASTY
IN CONGENITAL
NEUROGENIC
velopharyngeal closure the child still requires proper tongue and lip movement to complete articulation. All these children required speech therapy after surgery both to maximise their intraoral breath pressure and to try to eliminate poor articulatory habits. As Albery etal. (1982) suggest, “A successful operation . . merely prepares the ground on which the game of articulation must be played”. The duration of follow-up (Fig. 6) indicates the persistence required to achieve the best possible articulatory patterns. Our results suggest possible reasons for the successful outcome in a case of post-traumatic palatal paralysis (Crikelair et al., 1970) and express more precisely Heller et al.? (1974) comment that severe impairment of articulatory function is a contraindication to surgery. Successful surgery did not appear to depend on the child’s age or intellect in this small group, for although no formal IQ test was performed, it should be recognised that most of these children were referred by a neurologist who had excluded those of very low IQ as well as those with progressive diseases. Some authors have advocated palatal lift devices (Hardy et al., 1969; Gonzalez and Aronson, 1970; Selley, 1979), first described by Gibbons and Bloomer (1958). Their main advantage is the avoidance of the dangers and psychological trauma of surgery. They are also adjustable. The device consists of a dental plate fitted with an extension that elevates the velum directly. It requires sound teeth, which may be at risk with prolonged wear, and demands good oral hygiene. The growing child may not tolerate a plate needing frequent adjustment and replacement, particularly as local irritation is common in the initial stages. Heller et al. (1974), commenting on these limitations, believed that “the indications for surgery had been too conservative”. One preliminary report describes 2 cases with favourable speech changes following pharyngoplasty, in whom palatal lift devices had failed (Johns, 1982). Blakely (1964) has argued against long-term prostheses but suggests they have a role on a temporary basis, believing the prosthesis maximises muscle function preoperatively. The authors have no experience of the use of such devices in children with neurological speech disorders. A clear indication for this type of appliance is for the patient with respiratory muscle involvement, and other situations when surgery should be avoided.
195
SPEECH DISORDERS
In conclusion, this study suggests that pharyngoplasty has a definite role to play in the management of these unfortunate children. Neither the specific aetiology nor the type (spastic, flaccid, mixed) of their dysarthria appears important, but the severity of disability does influence the likelihood of speech improvement. Severe impairment of tongue and lip function, recognisable by simple observation, signifies a poor chance of success. Advancing and/or global neurological disease also represent contraindications to surgery. The effects of low intelligence are much more difficult to assess. Though our patients represent a spectrum of intelligence, none was grossly subnormal. The child’s hearing, maturity and co-operation, together with the degree of parental support, are other factors which need to be considered. Every child requires thorough investigation to confirm that velopharyngeal incompetence exists, to ascertain that it occurs continuously and not intermittently, to reveal any hidden anatomical defect as its cause and to enable the surgeon to design the most appropriate flap to obturate the defect. Our results show that when faced with this difficult clinical problem, unbridled pessimism is unwarranted. We would support the view of Randall et al. (1960) that even a minor improvement in intelligibility may lead to the most gratifying personality changes as the child gains in selfconfidence and self-respect. Acknowledgements The authors wish to thank Miss L. Skeates for her illustrations, Drs J. K. Brown, R. Minns and A. O’Hare of the Department of Paediatric Neurology, Royal Hospital for Sick Children in Edinburgh, for their help and advice in the preparation of the manuscript. Dr R. Prescott and Mr W. Adams from the Department of Medical Statistics, Edinburgh University, provided the statistical analysis.
References Ah-y, E. H., Bennett, J. A., Pigott, R. W. and Simmons, R. M. (1982). The results of 100 operations for velopharyngeal incompetence - selected on the findings of endoscopic and radiological examination. British Journal of Plastic Surgery, 35, 118. Blakely, R. W. (1964). The complementary use of speech prostheses and pharyngeal flaps in palatal insufficiency. Clefr Palate Journal, I, 194. Boorman, J. G. and Sornmerlad, B. C. (1987). Palate re-repairrationale, technique and results. Presentation at the British Association of Plastic Surgeons Winter Meeting. Broadbent,
T. R. and Swinyard,
C. A. (1959).
The dynamic
196 pharyngeal flap; its selective use and electromyographic evaluation. Plastic and Reconstructive Surgery. 23, 30 1. Brown, J. K. (1985). In Darley, J. K. (Ed) Speech and Language Evaluation in Neurology: Childhood Disorders. USA, Canada, England : Grune and Stratton, pp. 130-l 84. Crikelair, G. F., Kastein, S. and Cosman, B. (1970). Pharyngeal flap for post-traumatic palatal paralysis. Plastic and Reconstructive Surgery, 45, 182. Gibbons, P. and Bloomer, H. (1958). A supportive-type prosthetic speech aide. Journalof Prosthetic Dentistry, 8, 362. Gonzalez, J. B. and Aronson, A. E. (1970). Palatal lift prosthesis for treatment of anatomic and neurologic palatopharyngeal insufficiency. Cleft Palate Journal, 7,91. Hardy, J. C., Netsell, R., Schweiger, J. W. and Morris, H. L. (1969). Management of velopharyngeal dysfunction in cerebral palsy. Journal of Hearing and Speech Disorders, 34, 123. Heller, J. C., Gens, G. W., Moe, D. G. and Lewin, M. L. (1974). Velophamygeal insufficiency in patients with neurologic, emotional and mental disorders. JournalofHearing and Speech Disorders, 34, 350. Johns, D. F. (1982). Clinical Management of Neurogenic Speech Disorders. Boston: Little, Brown&Co., p. 167. Kaplan, E. N. (1975). The occult submucous cleft palate. Cleft Palate Journal, 12,356. Mmami, R. T., Kaplan, E. N., Wu, G. and Jobe, R. P. (1975). Velopharyngeal incompetence without overt cleft palate, a collective review and experience with 98 patients. Plastic and Reconstructive Surgery, 55, 573.
BRITISH JOURNAL
OF PLASTIC SURGERY
Pigott, R. W., Benson, J. F. and White, F. D. (1969). Nasendoscopy in the diagnosis of velopharyngeal incompetence. Plastic and Reconstructive Surgery, 43, 171. Randall, P., Bakes, F. P. and Kennedy, C. (1960). Cleft palatetype speech in the absence of cleft palate. Plastic and Reconstructive Surgery, 25,484. SeUey, W. G. (1979). Dental and technical aids for the treatment of patients suffering from velopharyngeal disorders. In Ellis, R. E. and Flack, F. C. (Eds) Diagnosis and Treatment of Palatoglossal Malfunction. London: College of Speech Therapists.
The Authors P. M. Davison, FRCS, Consultant Plastic Surgeon, North Staffs Royal Infirmary, Stoke-on-Trent; formerly Senior Registrar in Plastic Surgery, Royal Hospital for Sick Children, Edinburgh Ros E. RazzelI, LCST, Senior Speech Therapist A. C. H. Watson, FRCSEd, Consultant in Plastic Surgery Royal Hospital EH9 1LF. Requests
for Sick Children,
for reprints
to Mr Watson
Sciennes
Road,
Edinburgh
at the above address.
Paper received 28 April 1989. Accepted 31 August 1989 after revision.
The role of pharyngoplasty speech disorders P. M. DAVISON,
in congenital
neurogenic
R. E. RAZZELL and A. C. H. WATSON
Royal Hospital for Sick Children, Edinburgh Summary-Children with dysarthria are often seriously handicapped by their inability to communicate. Velopharyngeal incompetence may contribute to their speech difficulties but the indications for pharyngoplasty are poorly defined. The speech of 41 children, thought to have a defect of neurological origin and referred for possible pharyngoplasty, was assessed by a speech therapist, by nasopharyngoscopy and by multiview videofluoroscopy. These investigations revealed that 16 had velopharyngeal incompetence, with a neurological aetiology, and might be expected to benefit from pharyngoplasty. Independent assessment of the intelligibility of these 16 children before and after pharyngoplasty has been related to their preoperative oropharyngeal function. The majority benefited from surgery, some substantially. In particular, good lip and tongue function are shown to be necessary to achieve improved intelligibility. The roles of age, intelligence and aetiology in the success of pharyngoplasty are discussed.
It was shown by Randall et al. (1960) that pharyngoplasty can improve the speech of patients with velopharyngeal incompetence due to neurological disorder. However, few guidelines on the indications for surgery have since emerged. It has been suggested by Heller et al. (1974) that reducing nasal emission and improving nasality will not necessarily lead to better intelligibility, unless dysarthric speech patterns can be amended. To date, most authors when discussing the surgical management ofdysarthric speech have documented changes in voice quality rather than changes in speech intelligibility as recommended by Johns (1982). Few papers have attempted to relate these changes to the function of structures involved in articulation. The dual objectives of this study were (1) to investigate, by assessment of their intelligibility, whether pharyngoplasty can benefit the speech of dysarthric children; (2) by quantifying function of the pharynx, the velum, the tongue and the lips, to identify any factors which could lead to better patient selection.
investigation of speech problems neurogenic origin.
of suspected
Initial assessment
A thorough speech assessment, as detailed in Table 1, was carried out on each child by an experienced speech therapist (R.E.R.). Video recordings were made of conversational speech, and nasopharyngoscopy and multiview videofluoroscopy performed. The function of each of the structures involved in articulation, i.e. the lips, the tongue and the soft palate together with that of the lateral pharyngeal walls and the posterior pharyngeal wall, was quantified (Fig. 1). Velar, lateral pharyngeal and posterior pharyngeal wall movements were judged by a combination of endoscopy and videofluorosTable 1
Methods of assessment of speech and language
Receptive language Expressive language Articulatory movements Articulation Nasal escape
Material and methods
Nasal resonance
Over a 7 year period, 1979-1986, 41 children were referred to the Edinburgh Cleft Palate Clinic for
Nasal grimacing
187
and snorting
Reynell or Reynell or Imitation EAT Subjective scale Subjective scale Observation
PPVT APT
judgement,
S-pt
judgement,
3-pt
188
BRITISH JOURNAL LATERAL PHARYNGEAL
SOFT PALATE /
\
LIPS
SCORE 0,l
WALL
POSTERIOR PHARYNGEAL
OR 2 FOR EACH OROPHARYNGEAL
OF PLASTIC SURGERY
WALL
MOVEMENT
Fig. 1 Figure l-The Oropharyngeal
summation of the scores Movement Score
from
each of the five functional
copy. Lip function was assessed by asking the children to purse and spread their lips, and tongue mobility by observation of tongue elevation, depression and lateral movement. By awarding 2 for normal function, 1 for reduced rate or amplitude, and 0 for no function for each of these five elements, we arrived at a total that, for the purpose of this paper, we have termed an Oropharyngeal Movement Score with a maximum score of 10. Diagnosis and management ofpatients
Whilst not every child allowed all the investigations to be performed, enough information was gleaned to eliminate 17 patients for reasons shown in Table 2. Twenty-four children were found to have veiopharyngeal incompetence which was contributing to their dysarthria; one was lost to follow-up. The management of the other 23 is shown in Table 3. The 6 children whose velopharyngeal incompetence was found to be intermittent, detectable only during conversational speech but not on isolated sounds, were advised to have further speech therapy. None has since been referred back for reassessment. Another had severe respiratory problems making surgery ill-advised. The ages of the 16 children who had a pharyngoplasty and form the basis of this study ranged from 3 to 15 years (Fig. 2). All had undergone
elements
involved
in articulation
was termed
the
prolonged speech therapy prior to surgery. The causes of their dysarthria, diagnosed by a neurologist, are given in Table 4. The pharyngoplasty was performed on all but one of the children by the same surgeon (A.C.H.W.). The technique used was tailored to the patient’s endoscopic findings. When a large central incompetent port was visible, a posterior pharyngeal hap was used. This was based inferiorly if there was neither palatal nor pharyngeal wall movement, but a superiorly based flap was employed in the presence of large adenoids or if there was some movement of the lateral pharyngeal walls, Patients eliminated from the study as they did not have both velopharyngeal incompetence and a neurological cause Table 2
Palatopharyngeal disproportion Microform cleft palate Immaturity Dysarthric but no velopharyngeal
incompetence
1 2 6 8
The management of dysarthric children who had velopharyngeal incompetence
Table 3
Pharyngoplasty undertaken Unfit for pharyngoplasty Further speech therapy alone
16 1 6
THE ROLE OF PHARYNGOPLASTY
IN CONGENITAL
NEUROGENIC
189
SPEECH DISORDERS
Patient Age when Pharyngoplasty Advocated
Figure 2-The
patient’s
age when pharyngoplasty
was recommended.
in an attempt to place the flap at the level of this activity. A wide transverse port with some palatal and no lateral wall movement was obturated by modified Hynes/Orticochea lateral wall flaps (Table 5). It was recognised that, in the presence of poor movement of both soft palate and pharyngeal walls, it would be impossible to achieve velopharyngeal competence without obstructing the nasal airway, and no attempt was made to do this. The aim was a compromise; to reduce nasal escape to acceptable levels while allowing adequate nasal airflow. This was done by raising flaps of moderate size. After surgery all the children continued with their speech therapy. Postoperative
each patient’s pre- and postoperative speech as recorded on U’matic videotape, with the screen hidden. The items of speech were presented in a Table 4 The causes of dysarthria had velopharyngeal incompetence
in the children
Upper motor new-one Cerebral palsy Arrested hydrocephalus Articulatory dyspraxia Suprabulbar palsy Generalised hypotonia
2 2 2 1 1
Lower motor newone Isolated palatal paresis Moebius syndrome
5 3
who
assesment
Postoperative assessment was a repeat of the complete preoperative work-up, with a follow-up period ranging from 6 months to 3 years, with a mean of 19 months. In addition a comparison was made between pre- and post-treatment intelligibility, as judged by an independent five-person lay panel. The panel members were asked to grade
Table 5 The pharyngoplasty complications
techniques
used, and their
Technique
No.
Mujor complications
Superior pharyngeal Inferior pharyngeal Hynes/Orticochea
6 5 5
Haemorrhage
X 1
190
BRITISH JOURNAL
random order and the panel were asked to place each item on the following 4-point scale : unintelligible poorly intelligible comprehensible normal
= 1 =2 z=3 =4
these specific details were unobtainable preoperatively. Taken together, these results suggest that an effective obturator was constructed in most of the
For each patient the mean intelligibility score was calculated both pre- and postoperatively and the difference between these figures has been used to define the Improvement in Intelligibility as shown in Figures 5, 7 and 8. The significance of these improvements has been tested using the Spearman Rank Correlation analysis and is quoted in the text.
POST
F
Results Initial speech assessment showed that most of the children had delayed receptive and more severely delayed expressive language abilities. Their imitation of articulatory movements and their articulation patterns were poor. All bar one had audibly detectable airflow problems with abnormal resonance patterns; the exception had gross velopharyngeal incompetence visible endoscopically during speech, despite his nasal escape being inaudible. Nasal grimacing and snorting were not outstanding features of the group. A breakdown of each individual child’s results is shown in Table 6. It is interesting to note that most of the children with Moebius syndrome not only had 6th and 7th cranial nerve palsies as is generally recognised, but also had problems with even simple tongue movements. Three out of 5 children referred with an apparently isolated palatal paresis were also found to have significant restriction of tongue and lip mobility. Figure 3 shows a comparison of pre- and postoperative nasal escape (after the method of Boorman and Sommerlad, 1987) measured on a 5point scale by the speech therapist, for fourteen children. Figures in the shaded area would represent patients whose escape had deteriorated with surgery, those in the stippled area the 3 patients in whom no change was detected. The 11 patients in the upper right-hand corner had all improved (p >O.OOl), some substantially. Figure 4 compares nasal resonance before and after surgery. The improvement in both nasal escape and resonance in the 2 remaining children cannot be illustrated as
OF PLASTIC SURGERY
OPERATIVE
SEVERE
2 ii 0
MODERATE
I MILD
NONE
Fig. 3 Figure &There was a significant nasal escape after pharyngoplasty.
POST
(p=O.OOl)
improvement
in
OPERATIVE
HYPER
P
NORMAL
0
E
HYPO
Fig. 4 Figure &Nasal resonance was reduced by pharyngoplasty in some patients. (N.B. Only the numbers in the unshaded vertical represent the intended surgical result).
4f
6
3
Superior
Unintelligible+
8
Superior
5
7
H/O*
Palatal
Unintelligible Poor + normal
2
Superior
6
Palatal paresis
14
Palatal paresis
Comprehensible
6
Superior
I4
Unintelligible Comprehensible
I
Unintelligible
7
Palatal paresis
* Hynes/Orticochea
+ comprehensible
Comprehensible
3
Superior
poor
+ unintelligible
-+ normal
+ normal
+ unintelligible
+ comprehensible
No audible nasal escape, but gross velopharyngeal insufficiency seen on endoscopy
15
-+ comprehensible
Comprehensible
2
H/O*
Inferior
14
II
Moebius syndrome
Palatal paresis
paresis
10
Poor + comprehensible
6
Superior
30
10
6
17
8
24
33
18
36
28
poor
Unintelligible+
+ unintelligible
7
Unintelligible
4
24
6
I
Inferior
by reactionary
22
Length o~followup (mthl
Inferior
+ normal
Flap disrupted haemorrhage
Comments
6
Comprehensible
intelligibilit)
H/O*
H/O*
5
Generalised
4
4
hypotonia
Articulatory
Moebius syndrome
dyspraxia
Articulatory
Moebius syndrome
palsy
dyspraxia
Suprabulbar
5
8
Arrested hydrocephalus
4
4
H/O*
I1
Arrested hydrocephalus
Poor -+ normal
Unintelligible
3
-+ poor
Poor + comprehensible
4
Inferior
palsy
Inferior
Tongue and lip Pre- and postoperative score
I1
15
Articulator) movement score
Kernicterus
Cerebral
details
Pharvngoplasty technique
patient
Age f yr)
Individual
Diagnosis on referral
Table 6
192
BRITISH JOURNAL OF PLASTIC SURGERY
I
I
z
1
POST ;PERATl”E
a
3
IMPROVEMENT IN INTELLIGIBILITY (4 point scale)
Fig. 5 Figure SImprovement in nasal escape could not be related to improvement in intelligibility after surgery (Improvement is defined in the text).
children but that the narrowing of the nasopharynx was probably overdone in 3 patients. None of these suffered any resultant disabling symptoms. There was no obvious correlation between the improve-
POST w
OPERATIVE Y
m
iii
x 5
2
ii G
z
2 5
5
i
e z
UNINTELLIGIBLE
F POOR is
ment in nasal airflow obtained and the type of pharyngoplasty performed. The panel were found to give a remarkably consistent judgement in the change between each child’s pre- and postoperative speech, allowing a quantitative assessment of improvement in intelligibility to be made. Thus a child who had started with poorly intelligible speech and who achieved normal speech after operation, judged as a mean by the panel, was graded as having made a 2+ improvement in intelligibility. Figure 5 shows that the improvements in intelligibility could not be related to the reductions in nasal airflow (p=n.s.) that have been demonstrated. Intelligibility before and after surgery is shown in Figure 6, indicating that none was worse off for having a pharyngoplasty, even though one child’s flap was disrupted by a reactionary haemorrhage; this was the only major complication in the series. Eleven of the 16 children had improved (p = O.OOl),including 5 who achieved normal or near normal speech. Of the 7 children who started with totally incomprehensible speech, 4 could make themselves understood, even if with difficulty. Three children remained unintelligible. Age did not appear to influence the degree of improvement, but an obvious trend is apparent when improvement is collated with muscle function as measured by the preoperative oropharyngeal movement score (p = 0.01) (Fig. 7). An even more emphatic relationship is revealed if just the tongue and lip movement scores are compared with intelligibility (p < 0.01) despite the apparent crudeness of the assessment (Fig. 8). It is interesting to note that tongue and lip function was virtually unchanged by the operation in any of the children. The number of children with each diagnostic label is too small for any relationship to emerge between aetiology and improvement in intelligibility. It was noted that children with upper motor neurone conditions tended to have better control of their tongue and lips than those with lower motor neurone conditions.
kk!
0 w
Discussion
COMPREHENSIBLE
NORMAL
Fig. 6 Figure &Intelligibility by pharyngoplasty.
was significantly
(p=O.OOl)
improved
Speech is fundamental to the developing child’s ability to communicate and is therefore crucial to his behavioural, social and intellectual performance. Serious communication problems lead to reduced self-confidence and frequently to emotional disturbances. Conversational speech needs cerebration both to
THE ROLE OF PHARYNGOPLASTY
IN CONGENITAL
NEUROGENIC
SPEECH DISORDERS
193
Neurological disorders of speech may therefore be divided into three broad categories : -cerebral disturbance (dysphasia) -disorders of the larynx or its innervation (dysphonia) -motor disorders of the articulatory muscles (dysarthria).
12
0
15
3
OROPHARYNGEAL
6
78
P
MOVEMENT SCORE
Fig. I Figure ‘I-The improvement in postoperative intelligibility was related (p = 0.01) to the preoperative Oropharyngeal Movement Score.
understand the spoken word and to formulate and produce a reply in correct syntax. To vocalise this reply, the respiratory and laryngeal muscles produce a vibrating column of air which is articulated upon by the co-ordinated movement of muscles of the pharynx, the velum, the tongue and the lips.
1
. -
0
1
2
TONGUE AND LIP SCORE
Fig. 8 Figure&The best indicator (p < 0.01) of improved intelligibility was the preoperative assessment of lip and tongue function.
Pharyngoplasty can do nothing to influence dysphasia or dysphonia but has been used in the treatment of dysarthria when failure of velopharyngeal closure seems to be a significant factor in causing poor intelligibility. The term dysarthria encompasses a range of conditions which includes flaccid and spastic paralysis, rigidity and ataxia of the muscles of articulation, depending on the site of the neurological lesion. Articulatory difficulties in the child may be caused by a great number of conditions (Brown, 1985), ranging from localised peripheral nerve lesions to gross central nervous system disturbances resulting in disorders of control of articulation. Many pathological states in children are induced by conditions such as asphyxia and jaundice which may produce diffuse neurological damage leading to a global neurological deficit, resulting in a mixture of disorders such as dysarthria, deafness and mental retardation, each of which could be responsible for speech disorders. Moreover, as the muscles involved with feeding and articulation have developed phylogenetically from the laryngeal sphincter, and the nuclei responsible for the innervation of these muscles are closely approximated around the 4th ventricle, it is not surprising that disorders of respiratory control, phonation and swallowing often accompany dysarthria. Thus speech disturbance in a child may well be the result of a combination of dysphasia, dysphonia and dysarthria. This multifactorial aetiological picture leads to great difficulties in predicting the likely benefit of pharyngoplasty for any given dysarthric child (and makes statistical analysis difficult). Only a few studies have attempted to study the role of pharyngeal surgery in the management of patients with neurogenic dysarthria. The results have been variable. Whilst Broadbent and Swinyard (1959) suggested that patients with palatal paresis might be helped by pharyngoplasty, Randall et al. (1960) showed that a reduction in velopharyngeal insufficiency could improve the speech quality of some dysarthric children. In their study the
194 results in a small number of children with isolated and non-progressive palatal paresis varied from the dramatic to the disappointing. One child with associated neurological disorders of adjacent structures gained little benefit. Low intelligence was felt to be a relative contraindication to surgery. A considerable improvement in the speech of 50% of patients with cerebral palsy was reported by Hardy et al. (1969), but the reasons for failure were difficult to identify; neither intelligence nor the severity of neuromuscular involvement was believed to be a prime factor: “The only common variant in the unsuccessful cases . . . is a lack of intrinsic motivation”. Psychological and social readiness for speech improvement, together with parent co-operation, were factors that Minami et al. (1975) emphasised when reviewing their disappointing results in a group of patients of widely differing ages with palatal paresis. They advocated that patients with neurological disorders of neighbouring structures should be excluded from surgery. Heller et al. (1974), who were more enthusiastic about the place of surgery, also emphasised the contribution of both parental and patient co-operation to success. Though they found the neurological diagnosis was irrelevant, they believed the severity of disability and the presence of associated handicaps, together with limited intelligence, mitigated against improvement. In order that any conclusions be drawn from a study, accurate assessment is vital. The standard preoperative work-up used by this Unit enabled us to rule out suspected velopharyngeal incompetence in 8 dysarthric patients, and to identify 6 children whose incompetence was intermittent. Anatomical defects were recognised in 3 children, 2 of whom might well have been missed without nasopharyngoscopy (Kaplan, 1975). Many of the previous studies in this field predated the common use of this investigation (Pigott et al., 1969), with the result that some patients may have been incorrectly diagnosed. It is important that the cause of dysarthria is identified as accurately as possible by a neurologist, not only to prevent the surgeon operating on a patient with progressive disease but also to document the degree of involvement of other systems, such as the respiratory apparatus, as either factor could be a contraindication to surgery. It is interesting to note that, despite a thorough neurological investigation, our work-up revealed a wide range of variation in the involvement of articulatory
BRITISH JOURNAL OF PLASTIC SURGERY
muscles in children with the same diagnostic label. This fact and the small numbers must inevitably complicate any attempt to relate the aetiology of the dysarthria to the results. The intelligibility of all 16 children in this series who underwent surgery had ceased to improve despite prolonged speech therapy. It is known that these patients are unlikely to improve with time (Minami et al., 1975) but it is important to have maximised all the facets of articulation as far as possible, with speech therapy, before recommending surgery. No single surgical approach was adopted as the authors believe that the technique employed should be dictated by the endoscopic and radiographic findings. Seventy-five per cent. of the children had reduced nasal emission following pharyngoplasty, some considerably. Half of the children remained hypernasally resonant, 3 became hyponasal, with only 4 considered to have normal nasal resonance. The improvement in nasal airflow could not be correlated with the recorded changes in intelligibility. When viewed in isolation, it is not surprising that improvement in any one aspect of voice quality is meaningless as so many other factors may also influence speech in these disabled children. We agree with Johns (1982) that measuring changes in intelligibility, rather than voice quality, is of more value in assessing the benefits of surgery. An unbiased judgement of intelligibility was provided by an independent lay panel who had never met any of the patients concerned. In their opinion 11 of the 16 children had improved, a finding which the authors found encouraging. The question arises, “Could the children who did not benefit have been identified preoperatively?” Measurement of the oropharyngeal movement score shows a not unexpected trend-those patients with greater articulatory muscle function are more likely to benefit. To have adopted a cut-off score of 5 would have eliminated all those whose speech failed to improve but also 2 patients who profited from surgery. Evaluation of tongue and lip function alone revealed a clear division between successful and failed cases. No patient who scored less than 3 for these functions improved, while all those who scored 3 or more showed better intelligibility postoperatively. These assessments were easy to perform and to score. This finding can be explained because the operation is designed to influence the function of velum and pharyngeal walls but has no effect on tongue and lips. In the presence of effective
THE ROLE OF PHARYNGOPLASTY
IN CONGENITAL
NEUROGENIC
velopharyngeal closure the child still requires proper tongue and lip movement to complete articulation. All these children required speech therapy after surgery both to maximise their intraoral breath pressure and to try to eliminate poor articulatory habits. As Albery etal. (1982) suggest, “A successful operation . . merely prepares the ground on which the game of articulation must be played”. The duration of follow-up (Fig. 6) indicates the persistence required to achieve the best possible articulatory patterns. Our results suggest possible reasons for the successful outcome in a case of post-traumatic palatal paralysis (Crikelair et al., 1970) and express more precisely Heller et al.? (1974) comment that severe impairment of articulatory function is a contraindication to surgery. Successful surgery did not appear to depend on the child’s age or intellect in this small group, for although no formal IQ test was performed, it should be recognised that most of these children were referred by a neurologist who had excluded those of very low IQ as well as those with progressive diseases. Some authors have advocated palatal lift devices (Hardy et al., 1969; Gonzalez and Aronson, 1970; Selley, 1979), first described by Gibbons and Bloomer (1958). Their main advantage is the avoidance of the dangers and psychological trauma of surgery. They are also adjustable. The device consists of a dental plate fitted with an extension that elevates the velum directly. It requires sound teeth, which may be at risk with prolonged wear, and demands good oral hygiene. The growing child may not tolerate a plate needing frequent adjustment and replacement, particularly as local irritation is common in the initial stages. Heller et al. (1974), commenting on these limitations, believed that “the indications for surgery had been too conservative”. One preliminary report describes 2 cases with favourable speech changes following pharyngoplasty, in whom palatal lift devices had failed (Johns, 1982). Blakely (1964) has argued against long-term prostheses but suggests they have a role on a temporary basis, believing the prosthesis maximises muscle function preoperatively. The authors have no experience of the use of such devices in children with neurological speech disorders. A clear indication for this type of appliance is for the patient with respiratory muscle involvement, and other situations when surgery should be avoided.
195
SPEECH DISORDERS
In conclusion, this study suggests that pharyngoplasty has a definite role to play in the management of these unfortunate children. Neither the specific aetiology nor the type (spastic, flaccid, mixed) of their dysarthria appears important, but the severity of disability does influence the likelihood of speech improvement. Severe impairment of tongue and lip function, recognisable by simple observation, signifies a poor chance of success. Advancing and/or global neurological disease also represent contraindications to surgery. The effects of low intelligence are much more difficult to assess. Though our patients represent a spectrum of intelligence, none was grossly subnormal. The child’s hearing, maturity and co-operation, together with the degree of parental support, are other factors which need to be considered. Every child requires thorough investigation to confirm that velopharyngeal incompetence exists, to ascertain that it occurs continuously and not intermittently, to reveal any hidden anatomical defect as its cause and to enable the surgeon to design the most appropriate flap to obturate the defect. Our results show that when faced with this difficult clinical problem, unbridled pessimism is unwarranted. We would support the view of Randall et al. (1960) that even a minor improvement in intelligibility may lead to the most gratifying personality changes as the child gains in selfconfidence and self-respect. Acknowledgements The authors wish to thank Miss L. Skeates for her illustrations, Drs J. K. Brown, R. Minns and A. O’Hare of the Department of Paediatric Neurology, Royal Hospital for Sick Children in Edinburgh, for their help and advice in the preparation of the manuscript. Dr R. Prescott and Mr W. Adams from the Department of Medical Statistics, Edinburgh University, provided the statistical analysis.
References Ah-y, E. H., Bennett, J. A., Pigott, R. W. and Simmons, R. M. (1982). The results of 100 operations for velopharyngeal incompetence - selected on the findings of endoscopic and radiological examination. British Journal of Plastic Surgery, 35, 118. Blakely, R. W. (1964). The complementary use of speech prostheses and pharyngeal flaps in palatal insufficiency. Clefr Palate Journal, I, 194. Boorman, J. G. and Sornmerlad, B. C. (1987). Palate re-repairrationale, technique and results. Presentation at the British Association of Plastic Surgeons Winter Meeting. Broadbent,
T. R. and Swinyard,
C. A. (1959).
The dynamic
196 pharyngeal flap; its selective use and electromyographic evaluation. Plastic and Reconstructive Surgery. 23, 30 1. Brown, J. K. (1985). In Darley, J. K. (Ed) Speech and Language Evaluation in Neurology: Childhood Disorders. USA, Canada, England : Grune and Stratton, pp. 130-l 84. Crikelair, G. F., Kastein, S. and Cosman, B. (1970). Pharyngeal flap for post-traumatic palatal paralysis. Plastic and Reconstructive Surgery, 45, 182. Gibbons, P. and Bloomer, H. (1958). A supportive-type prosthetic speech aide. Journalof Prosthetic Dentistry, 8, 362. Gonzalez, J. B. and Aronson, A. E. (1970). Palatal lift prosthesis for treatment of anatomic and neurologic palatopharyngeal insufficiency. Cleft Palate Journal, 7,91. Hardy, J. C., Netsell, R., Schweiger, J. W. and Morris, H. L. (1969). Management of velopharyngeal dysfunction in cerebral palsy. Journal of Hearing and Speech Disorders, 34, 123. Heller, J. C., Gens, G. W., Moe, D. G. and Lewin, M. L. (1974). Velophamygeal insufficiency in patients with neurologic, emotional and mental disorders. JournalofHearing and Speech Disorders, 34, 350. Johns, D. F. (1982). Clinical Management of Neurogenic Speech Disorders. Boston: Little, Brown&Co., p. 167. Kaplan, E. N. (1975). The occult submucous cleft palate. Cleft Palate Journal, 12,356. Mmami, R. T., Kaplan, E. N., Wu, G. and Jobe, R. P. (1975). Velopharyngeal incompetence without overt cleft palate, a collective review and experience with 98 patients. Plastic and Reconstructive Surgery, 55, 573.
BRITISH JOURNAL
OF PLASTIC SURGERY
Pigott, R. W., Benson, J. F. and White, F. D. (1969). Nasendoscopy in the diagnosis of velopharyngeal incompetence. Plastic and Reconstructive Surgery, 43, 171. Randall, P., Bakes, F. P. and Kennedy, C. (1960). Cleft palatetype speech in the absence of cleft palate. Plastic and Reconstructive Surgery, 25,484. SeUey, W. G. (1979). Dental and technical aids for the treatment of patients suffering from velopharyngeal disorders. In Ellis, R. E. and Flack, F. C. (Eds) Diagnosis and Treatment of Palatoglossal Malfunction. London: College of Speech Therapists.
The Authors P. M. Davison, FRCS, Consultant Plastic Surgeon, North Staffs Royal Infirmary, Stoke-on-Trent; formerly Senior Registrar in Plastic Surgery, Royal Hospital for Sick Children, Edinburgh Ros E. RazzelI, LCST, Senior Speech Therapist A. C. H. Watson, FRCSEd, Consultant in Plastic Surgery Royal Hospital EH9 1LF. Requests
for Sick Children,
for reprints
to Mr Watson
Sciennes
Road,
Edinburgh
at the above address.
Paper received 28 April 1989. Accepted 31 August 1989 after revision.
