British Journal of Plastic Surgery (1990), 43,6X&32 @ 1990 The Trustees of British Association of Plastic Surgeons

ooO7-1226/90/0043-0676/$10.00

The effects of very early palatal repair on speech

M. COPELAND

Plastic Surgery Unit, Stoke Mandeville Hospital, Aylesbury. Bucks

Summary-One hundred subjects were evaluated to investigate speech intelligibility at age 5 following very early palate repair. Evaluation was achieved using a visual analogue. The effect of articulation, nasal resonance and nasal escape on intelligibility, and other factors which may affect results, such as fistulae, otitis media, cleft types and age of repair, are discussed. Results show that 87 subjects had acceptable speech and 13 had unacceptable speech. Nine subjects had poor articulation, 9 had moderate to severe nasal escape and 7 had moderate to severe nasal resonance. Only 6 subjects required pharyngoplasty.

One of the major aims of palate repair is to allow the development of normal speech. We want to know as soon as possible after repair whether the child can be easily understood by his family, his peers and by strangers; that is, is he intelligible?

Intelligibility is the ease and speed with which a listener can understand or retrieve a message communicated within the context of spontaneous speech. As dynamic speech comprises many ele- ments which include rate, volume, accent and prosody as well as articulation, nasality and nasal escape, speech results dependent on one or other of these features alone cannot reveal overall commu- nication effectiveness. The aim of this study therefore is to describe results following very early repair in terms of intelligibility and then describe the features of articulation, nasality and nasal escape separately as these may have the greatest influence on intelligibility (Moore and Somers, 1975).

The terms “acceptable” and “unacceptable” are used to describe the degree of intelligibility after using predefined methods of clinical assessment. It was felt that other factors such as fistulae, hearing, type of cleft, influence of therapy, secondary surgery and exact age of repair may affect speech results, and these are also described. No attempt was made to look at language development in this study.

Good speech results following very early surgery have been predicted (Kaplan, 1981; Dorf and Curtin, 1982). Pre-speech vocalisations or babbling occurs well before the age of 6 months and babbling normally accelerates around that age (Henningsson, 1989). Abnormal speech patterns can be heard in infants of 5 months (Dorf and Curtin, 1982) so that palate repair before this time should decrease the

possibility of abnormal patterns of sound produc- tion being established. However, comparison be- tween studies relating speech to palate repair is difficult. Different criteria are used to describe speech, variables are not similarly controlled and variations occur in timing of speech assessments. Small sample studies and those which do not identify age of speech evaluation or which assess speech after 7 years of age, after palatal revisions or in relation to only one element such as articula- tion, nasal resonance or nasal escape alone, must be viewed with some scepticism. The only margin- ally comparable study we could find relevant to our methodology was that of 249 subjects in which speech was assessed at age five (O’Riain and Hammond, 1982) and repair was undertaken before the age of 30 months. Results found that 68% had “acceptable” speech and 32% had “grossly defec- tive or unacceptable” speech.

Method

Subjects

One hundred and thirty-seven consecutive subjects in the early repair series undertaken by Mr S. N. Desai at Stoke Mandeville Hospital were followed up. The criteria for inclusion were designed to standardise the age of repair and the age of speech evaluation. All surgery was performed by one surgeon, thus excluding unknown variables which may occur if surgery is performed by different surgeons or when multiple procedures are used.

The subjects all had cleft palate repair before the age of 6 months (range 9-25 weeks, mean 16.4 weeks) and a speech evaluation at an average age of 4.11 years (range 3.8-6.3 years, mean 5.5 years).

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THE EFFECTS OF VERY EARLY PALATAL REPAIR ON SPEECH 677

Of the 137, 17 subjects were excluded from this study as the presenting factors are known to affect normal speech development. These were identified as known low intelligence, muhiple congenital deformities, known moderate or severe hearing loss or independently diagnosed severe speech and language difficulties (e.g. dyspraxia, elective mu- tism). Five subjects had repair later than 6 months and 15 subjects were not available at age of assessment. No attempt was made to delete subjects according to cleft type. Of the 100 subjects included in this study, 53 had unilateral cleft lip and palate, 17 had bilateral cleft lip and palate and 30 had cleft of the posterior palate. Wardill-Kilner (92 subjects) and Langenbeck (8 subjects) procedures were used as described by Desai (1983).

Procedure

Speech evaluation took place in one session on the same day as the 5-year clinical review. Each sample was rated by the reviewing speech therapist. Samples were recorded using a Sony TC525 tape recorder and Sony ECM-220 microphone. Samples included the Edinburgh Articulation Test (Anthony et al., 1971) a non-standardised test of articulation, a conversational sample based on picture description, and counting from 1 to 10. Further data were taken from speech therapists’ reports, case history entries and medical records. Intraoral examination at the time of assessment was used to describe the presence and approximate size of fistulae.

Rating

(1) Intelligibility Conversational samples were rated by using a visual analogue, i.e. a 10 cm line was drawn with polarised ratings of “very easy to understand” to “very difficult to understand” (Fig. 1). A mid-point on the

Fig. 1

Figure I-Visual analogue used to plot intelligibility.

scale was then used to identify two groups: Group 1 with acceptable speech who were easy or very easy to understand, and Group 2 with unacceptable speech who were difficult or very difficult to understand. Twenty-five random samples were also rated by a panel of two lay and one clinical assessors, not speech therapists.

(2) Articulation Each sample was assessed to identify the following: (a) No problems. (b) Immature features. These are characteristics

which may be due to immature, but normal, phonological development and having a stand- ard score of 85 or more on the Edinburgh Articulation Test (EAT).

(c) Very immature features. These characteristics are symptomatic of more severely delayed phonological development, having a standard score of less than 85 on the EAT.

(d) Atypical features. These were identified as lateralisations of sibilants, retractions of plo- sives/fricatives, and mixed features of both.

(e) Compensatory features. These were articula- tions associated with velopharyngeal insuffi- ciency and described by Trost (1981).

(3) Nasal resonance Each parameter was subjectively rated on a O-2 point scale :

0 No abnormal nasality 1 Minimal hyponasality or minimal hypernasality 2 Moderate to severe hypernasality

(4) Nasal escape Each parameter was subjectively rated on a O-2 point scale :

0 No nasal emission 1 Minimal nasal emission 2 Moderate to severe nasal emission

Results

Intelligibility

The therapist’s ratings were compared with the panel’s ratings. The product moment correlations between the four variables (i.e. rater and three panel assessors) were as follows :

Variable 1 2 3 2 0.77 3 0.84 0.81 4 0.91 0.83 0.81

678 BRITISH JOURNAL OF PLASTIC SURGERY

where variable 1 = rater and variables 2,3,4 = panel. All coefficients are significantly different from zero at the 0.1% level of probability.

Group 1. Eighty-seven subjects had acceptable speech. Of these, 58 were very easy to understand and 29 were easy to understand.

of one or other of these elements. Seven subjects had combined but minimal abnormal nasality and nasal escape and three had moderate to severe nasal escape only. None of the subjects in this group had moderate to severe hypernasality (Table 1).

Group 2. Thirteen subjects had unacceptable speech. Seven subjects were difficult to understand and six were very difficult to understand (Fig. 2).

Features afleeting intelligibility

Articulation

Group 2. Four subjects had either no abnormal nasality or nasal escape, or only minimal degrees of either element. Four subjects had either moderate to severe abnormal nasality or moderate to severe nasal escape. In 5 subjects, moderate to severe abnormal nasality occurred together with moderate to severe nasal escape (Table 1).

Group 1. Fifty-seven subjects had either no prob- lems or some of the immature features expected within the normal 5-year age population. Two subjects had very immature phonology, 11 realised fricatives laterally, 13 had retracted errors, and in 4 subjects errors were mixed (Table 1).

Factors which may a#ect results

Fistulae and hearing

Group 2. One subject had predominantly lateral errors, two had predominantly retraction errors and one had mixed errors. Four subjects had very immature phonology with additional atypical errors and 5 subjects had compensatory articulation. These 9 subjects were deemed to have poor articulation (Table 1).

The number of subjects with fistulae and those requiring insertion ofgrommets, and the percentage of these with acceptable speech (Group 1) or unacceptable speech (Group 2) are given in Table 2.

Cleft types The relationship between cleft types and speech results are shown in Table 3.

Age of repair Nasal resonance and nasal escape We compared degree of intelligibility with specific Group 1. Seventy-seven subjects had normal reso- age of palate repair and the pattern which emerged nance and no nasal escape or only minimal degrees is shown in Figure 3.

60

50

40

3r

2(

I(

(

ACCEPTABLE SPEECH Group 1 (N = 87)

very easy easy difficult very difficult to understand to understand to understand to understand

UNACCEPTABLE SPEECH GROUP 2 (N q 13)

Fig. 2

Figure 2--Intelligibility results as plotted on visual analogue.

THE EFFECTS OF VERY EARLY PALATAL REPAIR ON SPEECH 679

Table 1 Results of articulation, nasal resonance and nasal escape following very early palate repair

Abnormal resonance

Normal/minimal 93% Moderate/severe 7%

Nasal escape

Normal/minimal 91% Moderate/severe 9%

Articulation

Good/some errors 91”/, Poor 97;

Table 2 Relationship between fistulae, otitis media and speech results

No fistulae Fistula less than 2 mm2 Fistula between 2 mm2 and 5 mm’ Fistula greater than 5 mm2 Grommets inserted Combined fistula and grommets

Total no. of % in Group % in Group subjects I 2

58 91 9 29 83 17 11 73 27 2 100 0

52 83 17 29 79 21

N=lOO N=87 N=l3

Secondary surgery None of the 100 subjects had pharyngoplasty or any palatal revisions prior to assessment except one subject who had closure of a fistula.

Speech therapy Forty-two subjects had some speech therapy before assessment. Of these, 13 in Group 1 and 3 in Group 2 had treatment for 2 years. None of the subjects had undergone an intensive therapy course.

Discussion

Evaluation of intelligibility The use of a numerical rating scale in the assessment of dynamic speech leads to misinterpretation and does not allow for listener variables. The visual analogue as a subjective tool is a realistic method of measurement. Its terms of reference can be easily

Table 3 Relationship between cleft types and speech results

Cleft types Total no. of % in Group % in Group subjects I 2

Unilateral lip and palate 53 85 15 Bilateral lip and palate 17 88 13 Cleft palate only 14 79 21 Cleft soft palate 16 100 0

N=lOO N=87 N=13

interpreted and it allows some variation. This is necessary in any subjective assessment where agreement may differ in some degree whenever expectations of personal standards are or are not met.

Evaluation of speech was not undertaken at the age of 3 because the length of samples needed required good child co-operation within one session and because immature phonology can affect overall communication (Grunwell, 1988), a variable which would have occurred more frequently at a younger age. If evaluation is done at age 7 when intelligibility is said to plateau (Fletcher, 1978), results may be affected by other treatments such as long-term speech therapy and secondary surgical intervention. For instance, reassessment of Group 2 at this age revealed that, of the 100 subjects, 94 had acceptable speech and only 6 were difficult to understand. No subjects were very difficult to understand. More subjects had longer periods of speech therapy, and although no subjects had any palatal revisions, 6 had undergone Hynes pharyngoplasty by age 7.

Features afleeting intelligibility

Articulation In Group 1, when articulation errors did occur, they were fewer per subject, were lateral and retracted but the retractions occurred mainly in the hard palate area rather than in the velar, pharyngeal or laryngeal areas. Further investigation needs to be done into the relationship between the articula-

680

100

90

80

70

60

% 5o

40

30

20

10

0

N q 14

3 months

BRITISH JOURNAL OF PLASTIC SURGERY

0 Acceptable Speech B Unacceptable Speech q Unacceptable Speech with compensatory articulation

N q 51

4 months

Fig. 3

Figure 3-Bar graph indicating percentage incidence of degree of intelligibility and compensatory articulation with palate repair at 3,4,5 and 6 months.

tion age-related errors in this group and cleft palate media may have more effect on speech acquisition but it is known that there is a general tendency for and looked at the presenting factors of the 29 children up to the age of 5 to palatalise consonants subjects with these symptoms. It was found that (Anthony e? al., 1971). In Group 2 there were fewer although 6 had no articulation errors, half of all the lateralisations but more errors per subject, and these not in isolation but with other features such

subjects with lateral errors and the 2 subjects in Group 1 with moderate nasal escape were in this

as very immature errors. In this group, retractions group. We then looked at those subjects who occurred far more frequently. Compensatory artic- presented only with fistula. We found that most of ulations were only noted in this group and they the subjects with fistula less than 2 mm square and were associated with moderate to severe hypernas- ality in conjunction with moderate to severe nasal

10 of the 13 subjects who had fistula greater than 2 mm square were very easy or easy to understand

escape. Five of the 6 subjects requiring pharyngo- even though two of them exhibited the moderate plasty displayed all these combinations which are nasal escape mentioned above. This supports the well-known indicators of velopharyngeal insuffi- ciency.

view that fistulae are not likely to affect intelligibil- ity to a great degree.

Nasal resonance and nasal escape Looking at the subjects with otitis media, we

Abnormal nasality and nasal escape did not appear found only 17% with unacceptable speech at time

in Group 1 except as minimal or isolated in the of evaluation. This may be explained by the findings that cleft children have normal aerated ears before

presence of good articulation, whereas most of the subjects in Group 2 had some degree of abnormal

the age of 16 weeks (Too-Chung, 1983) and that

nasality/nasal escape and when combined with routine very early audiological monitoring to

poor articulation these features had a direct maintain hearing levels during the developing years

influence on degree of intelligibility. begins immediately after birth at lip and/or palate repair at this unit.

Factors which may affect results

Fistulae and hearing Cleft t,vpes

We felt that the combination of fistulae and otitis The cleft palate only group had the highest incidence of unacceptable speech (21%). The

N q 20

5 months 6 months

N=7

THE EFFECTS OF VERY EARLY PALATAL REPAIR ON SPEECH 681

subjects who presented with velopharyngeal insuf- ficiency had cleft of the palate only or cleft lip and palate ; none had bilateral clefts or soft palate only clefts. The two bilateral subjects in Group 2 had very immature phonology and retraction errors.

Age oj’repair The routine procedure was to time palate repair whenever possible at 4 months. Timing was not related to any other factor.

Henningsson (1989) reported that cleft palate infants appear to have a period of speech and language delay up to the time of repair, after which there is a rapid acceleration. Following Dorf and Curtin (1982) comparison between age of repair and speech results reveals a pattern which appears to indicate that palate repair at 4 months yielded the best results (Fig. 3), and although the numbers are far too small to draw any firm conclusions, it does emphasise the need for more research into the age at which cortical control over vocalisation begins and at what age repair can deter delay and physiological compensatory strategies from becom- ing too firmly established.

Secondary surgery All speech evaluations were done before pharyn- goplasty and the small number of pharyngoplasties is perhaps a good indication of the effects of very early repair on speech.

Speech therapy Speech therapy is a variable which cannot be ethically controlled. Therapy intervention must influence results; however, this study relates the amount of time over which therapy was given. The frequency patterns were typical normal practice and it is likely that similar levels of intervention would be found in any pre-school population of cleft palate children at the time this investigation was carried out although there are no statistics available to support this.

Although psychological aspects were not inves- tigated, they are worth comment. Many parents believe that major psychological benefits can be gained from very early lip and palate repair. The trauma of having a newborn baby with a facial deformity must be lessened to some degree if that deformity is corrected within hours of birth and before the baby is taken home. This must encourage early acceptance into the family and may reduce the low level expectations often described in studies of parental attitude (Richman and Eliason, 1982).

Motherchild bonding is not felt to be affected as the mother is with the baby during the first hours at lip repair and she is able to receive professional support and counselling at this very early stage. Very early palate repair may also minimise the disruptions and anxieties in the pre-school years that late repairs may cause, and may also encourage family integration and normality.

Conclusion

The main aim of this investigation was to evaluate the results of very early palate repair in terms of overall intelligibility.

Results show that at age five, 87 of the 100 consecutive subjects had acceptable speech. Of the 13 subjects who had unacceptable speech, 6 presented with symptoms of velopharyngeal insuf- ficiency. Of the total, 9 subjects had poor articula- tion, 7 had moderate to severe nasality and 9 had moderate to severe nasal escape.

Other factors which were felt may affect results were also described. Of these, specific age of repair revealed a pattern which appeared to indicate that very good results diminished gradually after the fourth month. However, much larger samples and more investigation into the age of onset of cortical control over vocalisation are required before firm conclusions can be made. Research is also needed to identify the psychological benefits which it is felt accompany very early lip and palate repair.

Acknowledgements

The author wishes to thank Dr L. Barr, Statistics Department, Oxford Regional Health Authority, and Mrs D. Gomm, Miss K. McCree and Mr J. Dickinson for their help with the assessments.

References

Anthony, A., BogIe, D., Ingram, T. and Mclsaac, M. (197 1). The Edinburgh Articulation Test. Edinburgh: Churchill Living- stone.

Desai, S. N. (1983). Early cleft palate repair completed before the age of 16 weeks: observations on a personal series of 100 children. British Journal ofPlastic Surgery, 36, 300.

Dorf, D. and Curtin, M. (1982). Early cleft palate repair and speech outcome. Plastic and Reconstktive &gery, fO.74.

Fletcher, S. G. 11978). Diunnosina Soeech Disorders from Cleii Palat;. New York:‘Grune and Stratton.

Grunwell, P. (1988). Phonological assessment, evaluation and explanation of speech disorders in children. Clinical Linguistics and Phonetics, 2, 221.

Henningsson, G. (1989). Cleft palate babbling related to time of palatal repair. In Kriens, 0. (Ed.) Proceedings of an -4dranced Workshop. Bremen 1987.

682 BRITISH JOURNAL OF PLASTIC SURGERY

Kaplan, E. (1981). Cleft palate repair at three months? Annaisof Plastic Surgery, 7, 179.

Moore, W. H. and Somers, R. K. (1975). Phonetic contexts: their effects on perceived intelligibility in cleft palate speakers. Folia Phoniatrica, 27,410.

O’Riain, S. and Hammond, B. N. (1972). Speech results in cleft palate surgery: a survey of 249 patients. British Journul of Plastic Surgery, 25,380.

Too-Chung, M. A. (1983). The assessment ofmiddle ear function and hearing by tympanometry in children before and after early cleft palate repair. British Journal of Plastic Surgery, 36, 295.

Richmao, L. C. and Eliason, M. (1982). Psychological character- istics of children with cleft lip and palate: intellectual, achievement, behavioral and personality variables. Cleft Palate Journal, 19,249.

Trost, J. E. (1981). Articulatory additions to the classical description of speech of persons with cleft palate. Cleft Palate Journal, 18, 193.

The Author

MrsMargaretCopeIand, ChiefSpeechTherapist, PlasticSurgery Unit, Stoke Mandeville Hospital, Aylesbury, Bucks HP21 8AL.

Requests for reprints to the author,

Paper received 13 October 1989. Accepted 28 May 1990 after revision.