Abstract
The purpose of this controlled study is to determine satisfaction with speech and facial appearance in Flemish pre-pubescent children with unilateral cleft lip and palate. Forty-three subjects with unilateral cleft lip and palate and 43 age and gender matched controls participated in this study. The Cleft Evaluation Profile was used to assess the perceived satisfaction for individual features related to cleft care. Both the cleft palate subjects and their parents were satisfied with the speech and facial appearance. The Pearson χ 2 test revealed significant difference between the cleft palate and the control group regarding hearing, nasal aesthetics and function, and the appearance of the lip. An in depth analysis of well specified speech characteristics revealed that children with clefts and their parents significantly more often reported the presence of an articulation, voice and resonance disorder and experienced /s/ /r/ /t/ and /d/ as the most difficult consonants. To what extent the incorporation of specific motor oriented oral speech techniques regarding the realisation of specific consonants, attention to vocal and ear care, and the recommendation of secondary velopharyngeal surgery, with the incorporation of primary correction of the cleft nose deformity simultaneously with primary lip closure, will aid these patients are future research subjects.
Two specialists who participate in the rehabilitation of patients with cleft lip and palate throughout childhood, adolescence and sometimes adulthood are the plastic surgeon and the speech–language pathologist. At least 50% of cleft palate patients require the services of a speech–language pathologist at some point in their lives . The specific treatment goals of speech language therapy are to establish correct articulatory placement, to modify resonance disorders and to treat accompanying language and voice disorders resulting in good overall speech intelligibility and satisfaction with speech. It is known that facial appearance and speech outcome may affect psychosocial functioning . Evaluation of the satisfaction of speech and facial appearance by the child and parents during and after multidisciplinary treatment in a craniofacial centre is a worthwhile topic in healthcare quality management. The primary goal of palatoplasty is to provide an intact hard and soft palate and to create a normally functioning velopharyngeal mechanism and communication profile. All other non-verbal indications for palate repair are secondary. When the velopharyngeal mechanism does not function adequately to prevent the transmission of sound into the nasal cavity, resonance disorders, a compensatory retracted articulation of anterior pressure consonants or fricatives, can occur. The success of the cleft palate surgery is specifically determined by the subsequent speech characteristics. The surgeon chooses the technique according to principles based on experience. Knowledge of the satisfaction of speech and facial appearance in subjects with cleft palate and their parents, whether or not in combination with objective outcome measures is interesting in order to change or ameliorate cleft palate related surgical procedures.
There are few studies clearly identifying specific parent and child reports regarding satisfaction with speech and facial appearance. Table 1 provides a summary of previous studies regarding the self-reports and/or parent reports of satisfaction of speech and facial appearance in the cleft palate population.
| Authors | Subjects ( n )/age | Type of cleft | Methods | Results |
|---|---|---|---|---|
| B roder et al. | * 495 parent–child pairs * Three age groups: 174: 5–9 years/124: 10–13 years/197:14–18 years * No control group * Patients from a large interdisciplinary university-based craniofacial treatment centre |
* CL ( n = 55) * CLP ( n = 235) * CP ( n = 205) |
* Interview | * 54% CL very pleased with appearance * 62% CP/CLP: very pleased with speech * Satisfaction with appearance: Significant correlation between female cleft subjects and parents * Older CL/CLP: increased satisfaction with speech * Parents of girls/boys more concerned about their daughters’ appearance/sons’ speech |
| T homas et al. | * 111 subjects with clefts * 62 parents * Three age groups: 10 years, 15 years or 20 years * Patients from nine clinics * No control group |
* UCLP ( n = 55) * CL ( n = 11) * BCLP ( n = 23) * CP ( n = 20) * SM ( n = 2) |
* Ratings of four features of facial appearance | * 20-year-old subjects significantly more satisfied with appearance than the 10 and 15 year olds * Subjects with visible clefts more dissatisfied than subjects with invisible clefts |
| M illard & R ichman | * 65 cleft palate children * No control group * Patients from one cleft palate clinic |
* UCLP ( n = 25) * BCLP ( n = 21) * CP ( n = 19) |
* Anxiety scale * Depression inventory * Interview |
* CP: greater problems with parent- and teacher-reported depression, anxiety, and learning related to speech than UCLP and BCLP * UCLP + BCLP: problems with facial appearance |
| M arcusson et al. | * 68 adults * Control group: 66 adults matched by sex and age * Patients from one department of plastic surgery |
* UCLP ( n = 46) * BCLP ( n = 12) |
* Self-report questionnaire | * More dissatisfaction with facial appearance (nose, lip, mouth, profile, overall facial appearance) with CLP * Dissatisfaction with facial appearance most significant predictor of depression * CLP group well-adjusted to disability |
| S inko et al. | * 70 adults * Age range: 18–30 years * Patients from one department |
* UCLP or BCLP | * Questionnaire on health-related quality of life | * 63% of the female patients want upper-lip and nose correction * Low scores for social functioning and emotional role, especially those patients who want further treatment |
| H unt et al. | * 160 subjects * Mean age: 13.6 years (8–21 years) *Control group: 13: 13 years (8–21 years) * Patients from three orthodontic departments |
* CLP | * Semi-structured interview for speech * Questionnaires |
* CLP greater behavioural problems, more depression, more teasing, less happy with facial appearance and speech * No significant difference between CLP and controls regarding anxiety or self-esteem * Teasing significant predictor of poor psychological functioning |
| N oor & M usa | * 60 (12–17 years) * No control group |
* CL ( n = 13) * CLA ( n = 2) * CLP ( n = 32) * CP ( n = 12) * SC ( n = 1) |
* Questionnaires * Interview |
* Teased about cleft related features such as speech, teeth and lip appearance affecting their self-confidence |
| H unt et al. | * 129 parents of children with CLP * Control group: 96 parents of children without CLP * Patients from three orthodontic departments |
* CLP | * Interview * Questionnaires |
* Child with CLP more teased, less satisfied with speech, less self-esteem, greater anxiety and unhappier compared with controls * Presence of CLP, appearance happiness, previous history of CLP in the family and visibility of scar affected parents’ ratings |
| O osterkamp et al. | * 43 (m.a.: 28.2 years) * Control group ( n = 43) matched for age, gender and socioeconomic status * Patients from one medical centre |
* BCLP | * Questionnaire | * Less satisfied with upper-lip, nose and nasal breathing * Satisfaction with facial appearance correlated positively with health-related quality of life * No difference for speech, hearing and drinking between cleft and non-cleft population |
| H avstam et al. | * 35 * Mean age: 25 years (22–32 years) * No control group * Patients from one cleft palate centre |
* UCLP ( n = 25) * BCLP ( n = 10) |
* Questionnaire | * Satisfaction with speech: high * No correlation between patients’ satisfaction with speech and professional evaluations * Men more dissatisfied with their speech * More women wanted corrections to their lips * Parents of girls/boys more concerned about their daughters’ appearance/sons’ speech |
| B erger et al. | * 145 adolescent-parent pairs * Between 11 and 16 years * Comparison with published normative data |
* CL ( n = 20) * CLP ( n = 71) * CP ( n = 54) |
* Questionnaires | * Adolescents/parents with visible cleft significantly less satisfied with appearance compared to a non visible cleft * Mothers more satisfied with their adolescent’s appearance than the adolescent themselves * Cleft group: less satisfied |
| M eyer -M arcotty & E isenhauer | * 3D facial data of 30 adults * Mean age 25.4 years |
* UCLP ( n = 24) * UCL ( n = 6) |
* Measuring 3D asymmetry (entire face + mid + -lower face) * Questionnaire |
* Asymmetry in the midface detract how facial appearance is self-perceived and perceived by others explaining the desire for nose correction |
Comparison of the methodology revealed that most studies used a questionnaire sometimes in combination with an interview and that several studies used no control group , a wide age range or patients were selected from different centres which makes interpretation of the results difficult. Contradictory findings were discovered. Whilst B roder et al. reported that more than 50% of the cleft palate subjects were very pleased with their appearance and speech, M illard & R ichman and M arcusson et al. concluded that there was more dissatisfaction with facial appearance in cleft palate subjects in comparison with the control group. N oor & M usa found that cleft palate children are teased, which affects their self-confidence, whilst H unt et al. reported no differences between the cleft palate group and the control group regarding self-esteem. Studies investigating satisfaction with speech and facial appearance in Flemish children as an outcome measure have not been performed to the best of the authors’ knowledge and are urgently suggested by M ossey et al. in their seminar of cleft lip and palate.
The main purpose of this study is to determine satisfaction with speech and facial appearance in Flemish pre-pubescent children with unilateral cleft lip and palate who require no more lip or palatal surgery or speech–language intervention at this time. All patients are treated by the craniofacial team of the University Hospital of Gent. Based on the results of previous reports in the literature a significantly greater satisfaction with speech and facial appearance was hypothesized in pre-pubescents with no clefts compared with pre-pubescents without a cleft. An additional objective of this study is to describe self-perceived speech characteristics and to delineate whether parents and children with clefts share similar perceptions regarding speech and appearance and if there are gender and age related differences.
Methods and materials
Forty-three subjects responded positively to an invitation to participate in this study. They ranged in age from 10 to 17 years (mean 11 years). The subjects were between 10 and 20 years, with non-syndromal unilateral cleft lip and palate, no secondary pharyngeal surgery, no cognitive deficiency, no neuromotor dysfunction or residual hard palate fistula and hearing thresholds less than 20 dB in the poorer ear. The selected subjects in the experimental group included 28 boys and 15 girls. All patients consulted the same craniofacial team and had undergone an identical surgical protocol. Surgical closure of the lip was performed using a modified Millard technique without primary nose correction at an average age of 5.4 months (range 3–5.11 months). The cleft had been closed using one-stage Wardill–Kilner palatoplasty at an average age of 13 months (range 11.8–23.8 months). All patients had been operated on by the same surgeon and with the same surgical technique in the same conditions. At an average age of 8.06 years (range 8–11.5 years), bone grafting (preceded by orthodontic treatment) was performed. Twenty-three children had speech therapy for a minimum of 8 months, twice a week. The goals of speech therapy were to establish correct phonetic placement and to eliminate compensatory articulations or developmental errors.
The gender- and age-matched control group were randomly selected and consisted of 43 children (38 boys, 15 girls) with a mean age of 12 years (range 7–18 years). All children went to normal schools and had no history or presence of ear, nose and throat pathologies. No significant differences in age were found between the boys and girls in the experimental group and those in the control group (ANOVA, P = 0.110). For this study each subject in the control and experimental groups was assessed by an otorhinolaryngologist who performed an examination to exclude nasal and ear pathologies using indirect laryngoscopy and macroscopic otoscopy. A Thudicum speculum was used to inspect the nose. Hearing was assessed for both ears separately at standard audiometric frequencies.
The Cleft Evaluation Profile (CEP) from the Royal College of Surgeons Cleft Lip and Palate Audit Group was used to assess the perceived satisfaction for individual features related to cleft care. An eight-item list regarding speech, hearing, lip, nose, teeth, breathing, and profile was used. The subjects and their parents were asked in an individual interview and in separate rooms to rate the satisfaction on a 4-point Likert scale ranging from very satisfactory (rank 1) to very unsatisfactory (rank 4). It is known that the CEP plays a major role in assessing facial appearance and speech amongst cleft lip and palate patients and was previously used to determine the perceived satisfaction of the patients and their parents with the clinical outcome of cleft treatment. The CEP is able to determine any significant differences in the parent and child ratings of the features that were related to speech and facial appearance. Additional information was asked in yes/no questions and open questions regarding specific speech characteristics. The researchers underwent training on how to administer the questionnaires to the patients in an unbiased manner to minimize intra examiner variability. The test-retest reliability of the CEP in the study of N oor & M usa showed it to be consistent in answering questions by both patients and parents. This study was approved by the ethical board (B67020096013) of the University Hospital Ghent, Belgium.
Statistical analysis
SPSS (V 18) was used for statistical analysis of the data. For the comparison of the nominal and ordinal data of the four groups the Pearson χ 2 test was used. For further analysis between the groups a Bonferoni correction was applied. A Pearson χ 2 test was employed to investigate gender and age (group 1, children younger than 12 years; group 2, children older than 12 years) related differences.
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