Longitudinal Evaluation of Articulation and Velopharyngeal

Transcription

1 _ Longitudinal Evaluation of Articulation and Velopharyngeal Competence of Patients with Pharyngeal Flaps D. R. Van Demark, PH.D. M. A. Harpin, PH.D. In this study, 129 patients with cleft palate who had pharyngeal flaps were evaluated longitudinally. Articulation scores improved approximately 15 percent following surgery, and by age 16 the majority of subjects achieved velopharyngeal competence and 90 percent correct articulation. The findings also demonstrated that the risk of requiring secondary management increased with the severity of the cleft. In addition, a higher percentage of patients who had received a primary Von Langenbeck procedure required secondary management than did patients who had the Wardill primary palatoplasty. Age at time of the pharyngeal flap operation did not emerge as a critical factor in speech outcome. It is generally reported that the pharyngeal flap operation successfully improves articulation and establishes velopharyngeal competence for approximately 80 percentof patients. At present, however, very little is known about the factors related to the success of the procedure. In an early study, Moll et al (1963) found that pharyngeal flap width and the patient's age at time of secondary operation were related to the success of the pharyngeal flap operation. More recently, Riski (1979) reported that patients who received pharyngeal flaps prior to 6 years of age demonstrated more rapid improvement in articulation and resonance balance than did children who received a pharyngeal flap after age 6. In contrast, Van Demark and Hammer- Dr. Van Demark is a Professor in the Department of Otolaryngology-Head and Neck Surgery and Department of Speech Pathology and Audiology at the University of Iowa, Iowa City, IA. Dr. Hardin is an Assistant Professor in the Department of Communication Sciences and Disorders at the University of Montana, Missoula, MT. This paper was presented at the Annual Meeting of the American Cleft Palate Association, Seattle, Washington, May 24, 1984, and was supported in part by Public Health Service Grant no. DE-00853, National Institute of Dental Research. 163 quist (1978) found that early secondary management (performed before 4 years of age) was not associated with better articulation proficiency or velopharyngeal competence. However, when secondary management was delayed until after 10 years of age, there was also a trend for poorer articulation. When compared at age 10, subjects who received a pharyngeal flap prior to 5 years of age were given poorer velopharyngeal competence ratings than subjects who received secondary management after 5 years of age. Van Demark and Hammerquist concluded that the optimal time of secondary management was between 4 and 10 years of age. Although the primary objective of their study was to examine the relationship between the timing of the pharyngeal flap operation and the success of the technique, Van Demark and Hammerquist also noted that type of cleft and type of palatoplasty appeared to be risk factors associated with the need for secondary management. According to the authors, subjects with cleft of the soft palate only, required a pharyngeal flap operation less frequently than subjects with more severe types of cleft. However, Riski's (1979) data indicated little difference in types of cleft that needed secondary management. Additional research is needed to iden-

2 164 Cleft Palate Journal, July 1985, Vol. 22 No. 3 tify those factors that are related to the success of the pharyngeal flap technique. Further study is also needed to identify those factors that may increase the need for secondary management. The purposes of this study were to examine the longitudinal development of articulation, nasality, and velopharyngeal competence of a group of subjects with cleft palate who had undergone the pharyngeal flap operation and had participated in the Iowa Cleft Palate Longitudinal Research Project. Specifically, the data, although necessarily retrospective in nature, could provide some insight into the following questions: 1) Does the age of initial palatoplasty appear to relate to the need of secondary management and speech results?» 2) Does the type of initial palatoplasty appear to relate to the need of secondary management and speech results? 3) Does the type of cleft appear to relate to the need of secondary management and speech results? 4) Does the age of pharyngeal flap secondary management relate to speech results? PROCEDURE Subjects All subjects in this study were part of the Iowa Cleft Palate Longitudinal Project. Initially, subjects were enrolled in the project when treatment was needed; however, after 1963 subjects were enrolled in the longitudinal project at birth. Approx- imately 520 subjects have been enrolled inthe project, which includes computer storage of pertinent speech-articulation data and information about various types of physical management. All subjects who had a secondary palatal procedure, which in this case was a superiorly-based pharyngeal flap operation, were identified from the computer identification program. A total of 129 subjects were available for study. Subjects' birthdates ranged from 1946 to 1975, with the mean year of birth, Information about the type of cleft and type of palatal operation for these subjects is shown in Table 1. Eleven of the 129 subjects had cleft of the soft palate only, 28 had cleft of the hard and soft palate, 58 had unilateral cleft of the lip and palate, and 32 had bilateral cleft of the lip and palate. As shown, the majority of subjects received either the Von Langenbeck or Wardill procedure for surgical repair of the palate. Timing of palatoplasty ranged from 3 months to 9 years of age, with a mean age of 3 years. Age of secondary management ranged from 2 years, 5 months to 16 years, 2 months, with a mean age of pharyngeal flap management of 7 years, 2 months. DATA COLLECTION Annual articulation test scores, obtained with the 105-item articulation test used in the research protocol, were retrieved for each subject using the computer program described previously by Van Demark and Tharp (1973). This test includes the Iowa Pressure Articulation Test, the Templin- Darley Screening Test of Articulation, as well as other phonemes in various word TABLE 1. Subjects Categorized According to Cleft Type and Type of Primary Palatoplasty Type of Cleft Male Female N Type of Surgery N Soft Palate Only (including subjects with submucous) l-stage Von Langenbeck 49 Hard and Soft Palate stage Von Langenbeck 14 Unilateral Lip and Palate Wardill (NVB * intact) 37 Bilateral Lip and Palate Wardill (NVB* severed) 24 Other 5 *NVB = neurovascular bundle

3 _ Van Demark and Hardin, LONGITUDINAL EVALUATION 165 positions. A total of 1506 articulation tests for the 129 subjects was available for study. Clinical ratings of velopharyngeal competence assigned by the examining speech pathologist during each examination were also retrieved. These clinical ratings (competence, marginal competence, or incompetence) were made by the speech pathologist as the best estimate of the adequacy of the mechanism. Routinely, these ratings were based on a combination of observations including conversational speech, word, and sentence articulation testing, stimulability, examination of the oral mechanism, and for some patients, information obtained from lateral x-ray films and fluoroscopy. Parental reports and reports from speech clinicians working with the child locally were considered in the composite rating. In addition, severity ratings (7 point scales where 1 = normal and 7 = severe) of nasality and articulation defectiveness in connected speech were retrieved. Approximately 90 percent of the observations obtained from the longitudinal project were made by two speech pathologists who were experienced in evaluating the speech of children with cleft palate. Estimates of their current interreliability in articulation test scoring and severity ratings of articulation and nasality were obtained in a recent report (Hardin, 1984). Agreement between the two examiners in scoring articulation tests was high (84%). Interjudge reliability of severity ratings was also relatively high with ratings exact or within one scale value on 92 percent (articulation) and 84 percent (nasality) of the samples. An estimate of their interjudge reliability in assigning clinical judgments of velopharyngeal competence was obtained in an earlier study (Morris, 1978), and 84 percent agreement was reported. RESULTS The articulation test scores were examined prior to and following the pharyngeal flap operation. As demonstrated in Table 2, the articulation scores before secondary management were poor. Subjects between the ages of 4"/;, and 8 years im- proved their articulation scores by approximately 15 percent (average difference in pre and post scores) following secondary management. After age 8, the articulation test scores of the pre- and postpharyngeal flap groups were more similar. It should be noted that the number of subjects gets smaller across age levels for the pre-flap group because as age increases, more subjects received pharyngeal flaps. Conversely, for the post-flap group the number of subjects becomes larger because additional subjects had pharyngeal flap operations at each age level studied. Although some individual subjects achieved relatively high articulation scores, the group data indicate that the articulation performance of these subjects was well below that expected of 8-year-old children without clefts and slightly below the norms of children with clefts (Van Demark et al, 1979). The most frequent error types observed prior to secondary management included omissions, sound substitutions, and nasal distortion of consonant sounds (Table 3). Although the number of errors related to omissions and substitutions decreased with age, nasal distortion errors did not. Analysis of the data following pharyngeal flap management revealed a substantial decrease in articulation errors related to na- TABLE 2. Mean Percentage of Elements Correct on the 105-Item Iowa Articulation Test Prior to and Following the Pharyngeal Flap Operation Age N* Before Flap Nt After Flap / *Ns decrease in column 2 because patients received pharyngeal flaps at approximately the age level indicated. {Ns increase in column 4 because of the increase in the number of subjects with pharyngeal flaps.

4 ' 166 Cleft Palate Journal, July 1985, Vol. 22 No. 3 TABLE 3. Mean Percentage of Elements Misarticulated on the 105-Item Iowa Articulation Test According to Type of Error for all Subjects Oral Nasal Oral Nasal Age N Distribution Distribution Substitution Substitution Omission Pre* Post* pre post pre post pre post pre post pre post / *Pre = pre flap, Post = post flap TABLE 4. Mean Percentage of Elements Correct on the 105-Item Iowa Articulation Test Following the Pharyngeal Flap Operation for all Subjects Grouped According to Timing of Primary Palatoplasty A <22 months months months months >46 months 8 N % N % N % N % N % / / sal distortion and an increase in oral distortion errors. Timing of initial palatoplasty was also investigated in order to assess the relation between the age at which palatoplasty was performed and articulation proficiency for patients with pharyngeal flaps (Table 4). Although the number of subjects in the early palatoplasty group (under 22 months) and the older palatoplasty group (over 46 months) was small, inspection of the data indicated that no readily apparent trend existed. The younger palatoplasty groups (under 22 months, 22 to 28 months, and 29 to 34 months) exhibited slightly superior articulation skills, correctly producing at least 80 percent of the test items by age 10. The two older groups did not reach that level of proficiency until 12 years of age. At age 12, however, the five groups were similar in speech proficiency with average scores of 87, 89, 85, 80, and 80 percent respectively. These findings suggest that timing of primary palatoplasty is not a critical factor in articulation proficiency for patients who require a pharyngeal flap procedure. Comparison of articulation performance between primary palatoplasty groups is shown in Table 5. In general, higher articulation scores were obtained by the Wardill group than the Von Langenbeck group following pharyngeal flap management. This trend is particularly noticeable until age 8, when the Von Langenbeck group performance was similar to the Wardill group. Table 6 illustrates the relation between articulation performance and the type of cleft. Subjects with cleft of the hard and

5 Van Demark and Hardin, LONGITUDINAL EVALUATION 167 TABLE 5. Percent Elements Correct on the 105- Item Iowa Articulation Test Following the Pharyngeal Flap Operation for all Subjects Grouped According to Type of Palatoplasty A Von Langenbeck Wardall 8 N % correct N % correct _ '/ / soft palate exhibited articulation skills slightly superior to subjects with cleft of the soft palate only; however, the differences were small and probably not meaningful because of the small sample size in both groups. Articulation proficiency between the groups with unilateral and bilateral clefts showed a slight tendency for subjects with unilateral clefts to have the better scores longitudinally. A phoneme analysis was performed for all subjects following secondary management to determine if differences existed between the group with cleft palate and the group with cleft lip and palate in the production of any consonant sounds. Although these data are not presented in this paper, subjects with cleft lip and palate exhibited substantially more difficulty than subjects with cleft palate only in producing alveolar sounds, particularly the /s/ and /z/ sounds. More errors might be expected for the groups with cleft lip and palate if dentition is a factor in articulatory production. Table 7 illustrates the relation between age at time of pharyngeal flap operation and articulation proficiency. Although the sample size of the youngest group (under 48 months at time of operation) is small, comparison of this group to the older age groups does not suggest a trend for better articulation with early secondary management. Subjects who had secondary management between 48 and 60 months tended to achieve slightly higher articulation scores than the youngest group and the oldest group at most age levels. This difference was small, however, and close inspection of the data indicates that the absolute difference between the group with the best articulation score and the group with the poorest score decreased with age. Although the older groups may exhibit poorer articulation skills immediately following pharyngeal flap management than the younger age groups, rapid improvement in articulation proficiency appears to occur. By age 12, all age groups were within approximately six percentage points of each other. Thus, our data are not supportive of the hypothesis that early secondary management (before age 4) results in higher articulation proficiency. TABLE 6. Mean Percentage of Total Elements Correct on the 105-Item Iowa Articulation Test Following the Pharyngeal Flap Operation for Subjects grouped According to Cleft Type. a spo H SSP ULP BLP 8 N % N % N % N % }, SPO = soft palate only, H&SP = hard and soft palate, ULP = unilateral lip and palate and BLP = bilateral lip and palate

6 168 Cleft Palate Journal, July 1985, Vol. 22 No. 3 TABLE 7. Mean Percentage of Elements Correct on the 105-Item Iowa Articulation Test for all Subjects Grouped According to Age at Time of Pharyngeal Flap Operation A <48 months months months months months >96 months N- % ON _ON % % _ / g TABLE 8. Risk Rates of Pharyngeal Flap for Iowa Patients Grouped According to Cleft Type and Type of Primary Operation Von Langenbeck Wardill Total Cleft Type Youd Patients Risk Tool Paliedts Risk Risk Patients with Flaps Patients with Flaps. : Soft palate Only % % 16% Hard and soft palate % % 31% Unilateral lip and palate % % 35% Bilateral lip and palate % % 39% Totals % % 32% RISK FACTOR FOR SECONDARY SURGERY Of particular interest was the significance of cleft type as a risk factor for requiring pharyngeal flap management. Risk was defined as the number of subjects with a specified cleft type who received a pharyngeal flap operation divided by the total number of subjects with that cleft type. The number of patients who have participated in the longitudinal cleft palate research project at Iowa is shown in Table 8 and is grouped according to the type of cleft and type of primary management. Also shown is the number of these patients who have received a pharyngeal flap operation. The results of our analysis suggest that risk of needing a pharyngeal flap operation is higher for patients with cleft of the lip and palate than for patients with cleft of the palate only. Patients with bilateral clefts of the lip and palate are at higher risk of requiring additional management than those with a unilateral cleft of the lip and palate. The risk of requiring secondary management is higher with clefts of the hard and soft palate than with clefts of the soft palate only. When the type of operation is considered for all types of cleft, it becomes evident that fewer subjects who received the Wardill procedure are in need of secondary management compared to those who had the Von Langenbeck procedure (26% versus 43%). The data in Table 8 illustrate that one should question using the Von Langenbeck procedure, particularly for patients withclefts involving the hard and soft palate and bilateral lip and palate. These differences, however, may reflect the fact that Von Langenbeck subjects generally had primary management before 1968,

7 Van Demark and Hardin, LONGITUDINAL EVALUATION 169 TABLE 9. Mean Rating of Articulation Defectiveness for all Subjects Grouped According to Age at Time of Pharyngeal Flap Operation Ave <48 months months months months months >96 months C_ w x N x N X N x N x N x / TABLE 10. Mean Rating of Severity of Nasality for all Subjects Grouped According to Age at Time of Pharyngeal Flap Operation. A <48 months months months months months >96 months W x- N N x N x N x N x '/g o o \ o while the Wardill subjects had primary management after that time. RELATIONSHIP OF SECONDARY MANAGEMENT TO ARTICULATION DEFECTIVENESS, NASALITY, AND VELOPHARYNGEAL COMPETENCE Clinical ratings of articulation defectiveness and nasality were also examined for subjects grouped according to age at time of secondary management (Tables 9 and 10). As shown, improvement in ratings of articulation and nasality occurred for all groups. Although the number of subjects in each management age group was small, a trend for better ratings of articulation was observed for the groups who received early pharyngeal flap operations (under 48 months; 48 to 60 months). Little difference between the groups was apparent in the severity of nasality. Clinical judgments of velopharyngeal competence were examined longitudinally for all subjects grouped according to the age at the time of pharyngeal flap management and are presented in Table 11. Again, little difference in average judgments of velopharyngeal competence was evident among the groups. These group data suggest that timing of secondary management is not an important factor in the outcome of velopharyngeal status. Additionally, the success of the pharyngeal flap procedure was evaluated, in part, by identifying the number of subjects who demonstrated velopharyngeal competence at the time of the last examination. Of the 129 patients, 102 received clinical judgments of velopharyngeal competence. Twenty subjects were judged to demonstrate marginal velopharyngeal competence and only seven subjects received

8 170 Cleft Palate Journal, July 1985, Vol. 22 No. 3 TABLE 11. Mean Rating of Velopharyngeal Competence for all Subjects Grouped According to Age at Time of Pharyngeal Flap Operation. A <48 months months months months months >96 months 6 N % ON- % No- % No- % No- 4% = velopharyngeal competence, 2 = marginal velopharyngeal competence, 3 = velopharyngeal incompetence judgments of velopharyngeal incompetence. Six of the 129 subjects exhibited consistent hyponasality following the pharyngeal flap procedure. Hyponasality was evident 1 to 2 years following secondary management for two subjects, 5 to 7 years for three subjects, and 11 years after secondary management for one subject. Three of the six subjects exhibited hyponasality for 2 consecutive years and three subjects exhibited it for 3 consecutive years. During their last examination, only two subjects were judged to have hyponasal voice quality. Likewise, information about mouth breathing was available for 114 of the 129 subjects at their last examination. Of the 114 subjects, 55 reported that they were mouthbreathers, and 48 subjects were identified by the examining clinician as mouth breathers. DIscUssION The results of this study indicate that one can expect improvement in articulation following pharyngeal flap management. Prior to the pharyngeal flap operation, subjects seldom, if ever, reached the mean correct score on the Iowa Pressure Articulation Test for cleft palate subjects (Van Demark et al, 1979); after a pharyngeal flap operation, differences in performance between flap and nonflap subjects of approximately 15 percent existed for most age groups. Differences were smaller at the older age levels, probably because most of the older subjects generally exhibited a marginal velopharyngeal mechanism and thus often had fairly acceptable articulation but unacceptable nasality. It should be noted that, as a group, articulation by the older subjects is still not completely normal and, as with subjects without flaps, speech habilitation is less than optimal (Bardach et al, 1984). Element analysis of articulation scores appears to us to be meaningful in the diagnostic management of individuals. It has been well documented that articulation errors occur most predominately on pressure sounds, plosives, fricatives, and affricatives, although fricatives and affricatives are learned relatively late and thus may not be differentially diagnostic. However, type of error may be more meaningful and merits further investigation. Normal children seldom exhibit nasal distortions or emission of air on pressure sounds. The inability of children with clefts to change this type of error over time may be the most significant cue in determining the need for further management. Our data demonstrate that there is a decrease in the number of nasal distortions and an increase in the number of oral distortions after pharyngeal flap management. Our data are in disagreement with those of Riski (1979) who found an approximately equal proportion of subjects with clefts of the palate versus clefts of the lip and palate who required flaps. Our data indicate that there is a greater risk of requiring secondary management for pa-

9 _ Van Demark and Hardin, LONGITUDINAL EVALUATION 171 tients with bilateral clefts, with the least risk for patients with clefts of the soft palate only. The severity of the cleft may influence the age at initial palatoplasty. In our institution, subjects with more severe clefts tended to have later management, and it is not clear that age at time of initial palatoplasty is a primary factor influencing the need for pharyngeal flap management. Thus, even with a relatively large sample size, it is difficult to sort out the influence of the type of cleft, type of primary surgical management, and age of primary operation on the need for secondary management. Review of the literature still indicates some confusion as to which operative management procedure gives better results. Overall, our data indicate superior results with the Wardill procedure; however, some caution should be exercised in the interpretation of the results since our Wardill subjects are younger. Some of these subjects may need secondary management in their teenage years. The data in Table 7 suggest that one of the reasons for inconsistency of various reports may be the make-up of the sample. It can be seen that differences in risk between the two types of palatoplasty are very small for subjects with cleft of the soft palate only and unilateral cleft lip and palate. The Von Langenbeck group, however, demonstrates a much greater risk of requiring secondary management for patients with clefts of the hard and soft palate and bilateral clefts. In our original research (Van Demark and Hammerquist, 1978), a slight difference was found in favor of the Von Langenbeck procedure for clefts of the soft palate only. It would appear that if the Von Langenbeck procedure is used, it should only be used for clefts of the soft palate and unilateral cleft lip and palate. Another factor which may influence these results is the age at the time of initial palatoplasty. On the whole, Von Langenbeck subjects had primary management later than the Wardill subjects. Data from this study suggest that the age at which the pharyngeal flap procedure is performed is not extremely critical; however, differences in the success of this procedure may be noted at age extremes. Our initial results (Van Demark and Hammerquist, 1978) indicated that subjects who received pharyngeal flaps before 4 years of age achieved poorer velopharyngeal competence; thus, the number of subjects in this study having a pharyngeal flap operation before this age is small. Because of the longitudinal nature of the project, few subjects had obvious velopharyngeal incompetence at the older age levels (1e., after 8 to 9 years of age). The majority of subjects who received secondary management after 8 years of age had marginal mechanisms prior to pharyngeal flap management, and they usually exhibited hypernasality but fairly good articulation, i.e., little audible nasal emission or compensatory articulatory behavior existed. An increase in the severity of hypernasality and audible nasal emission, probably related to adenoid atrophy, had become apparent for some subjects. Thus, as is demonstrated in Table 11, the age at time of pharyngeal flap management does not appear to be a significant factor in achieving adequacy of velopharyngeal competence. Likewise, the articulation scores, severity of articulation defectiveness, and severity of nasality appear highly similar, with a trend for subjects with early management to have slightly better scores. Recently, attention has been given to the detrimental effects of pharyngeal flap management, particularly in regard to nasal airway obstruction and sleep apnea. Forty-eight of our subjects were observed to be mouth breathers, a surprisingly high number. Unfortunately, we have not consistently observed the number of individuals with clefts who do not have flaps who exhibit a mouth-breathing problem, but we suspect that the incidence is relatively high and may result, in part, from septal deviations. Thus, patients tend to breathe through the pathway of least resistance, especially when involved in activity that requires physical exertion. REFERENCES BAarpACH J, MoRRIS HL, OLn WH, McDERMOTT- Murray J, Moony MS, BARDACH E. Late results of multidisciplinary management of unilateral cleft lip and palate. Ann Plast Surg 1984; 12:235.

10 172 Cleft Palate Journal, July 1985, Vol. 22 No. 3 HARDIN MA. The contribution of selected variables to the prediction of speech proficiency for adolescents with unilateral cleft of the lip and palate. Ph.D. Thesis, Iowa City: University of Iowa, MoLL KR, HUrFMAN WC, LIiErRLE DM, SMITH JK. Factors related to the success of pharyngeal flap procedures. Plast Reconstr Surg 1963; 32:581. MORRIS HL. Velopharyngeal competence and the Demjen W/V-Y technique: In: Morris HL, ed. The Bratislava project. Iowa City: University of Iowa Press, RiskI JE. Articulation skills and oral-nasal resonance in children with pharyngeal flaps. Cleft Palate J 1979; 16:421. Van DEMARK DR, THARP R. A computer program for articulation tests. Cleft Palate J 1973; 10:378. Van DEMARK DR, HAMMERQUIST PJ. Longitudinal evaluation of articulation and velopharyngeal competency of patients with pharyngoplasties. A paper presented to the American Cleft Palate Association, Atlanta, Georgia, VAN DEMARK DR, MORRIS HL, VANDEHAAR C. Patterns of articulation abilities in speakers with cleft palate. Cleft Palate J 1979; 16:200.