Incidence of cleft-related speech problems in children with an isolated cleft lip

Objectives

Clinicians agree that children with isolated cleft lip have fewer cleft-associated problems than children with cleft lip and palate. Unfortunately, for isolated cleft lip children, the risk of cleft-associated problems is unknown and maybe underestimated. Often, these children do not get the required follow-up by a multidisciplinary team and thereby not the known benefits in supporting their development. This study examines the incidence of cleft-related speech problems and ear problems in children with isolated cleft lip.

Materials and methods

A prospective study was performed on all children born with an isolated cleft lip and treated at the Wilhelmina Children’s Hospital in Utrecht between January 2007 and April 2014. Data were collected for sex, date of birth, genetics, cleft lip type, date of cleft lip repair, type of repair, speech/language problems, and ear problems.

Results

This study included 75 patients (59% male). The mean age of the children at the moment of speech examination was 32.5 months (SD 6.1). Eighteen of the 75 children (24%) needed speech and language therapy; however, only one child (1.3%) had a cleft-related speech problem. Sixteen of the 75 patients (21%) reported a history of one or more episodes of acute otitis media (AOM)/otitis media with effusion (OME) during the first 6 years.

Conclusion/clinical relevance

This is the first prospective study analyzing the incidence of cleft-related speech problems in children with an isolated cleft lip. These children do not have a higher risk of cleft-related speech problems or AOM/OME when compared to the general population. However, children with an isolated cleft do have a higher incidence of speech therapy.

     

Self-reports of psychosocial functioning among children and young adults with cleft lip and palate.

OBJECTIVE: A cross-sectional study was employed to determine the psychosocial effects of cleft lip and/or palate among children and young adults, compared with a control group of children and young adults without cleft lip and palate. PARTICIPANTS: The study comprised 160 children and young adults with cleft lip and/or palate and 113 children and young adults without cleft lip and/or palate. All participants were between 8 and 21 years of age. OUTCOME MEASURES: Psychological functioning (anxiety, self-esteem, depression, and behavioral problems) was assessed using validated psychological questionnaires. Happiness with facial appearance was rated using a visual analog scale. Social functioning, including experience of teasing/bullying and satisfaction with speech, was assessed using a semistructured interview. RESULTS: Participants with cleft lip and/or palate reported greater behavioral problems (p < .001) and more symptoms of depression (p < .01); they were teased more often (p < .001) and were less happy with their facial appearance (p < .01) and speech (p < .001), compared with controls. There were no significant difference between subjects with cleft lip and/or palate and subjects without cleft lip and/or palate in terms of anxiety (p > .05) or self-esteem (p > .05). Having been teased was a significant predictor of poor psychological functioning, more so than having a cleft lip and/or palate per se (p < .001). CONCLUSIONS: Teasing was greater among participants who had cleft lip and/ or palate and it was a significant predictor of poorer psychosocial functioning. Children and young adults with cleft lip and/or palate require psychological assessment, specifically focusing on their experience of teasing, as part of their routine cleft care.     

Dental development of Chinese children with cleft lip and palate.

OBJECTIVE: To compare the dental development and rate of tooth development between children with and without cleft lip and palate. Patients: Age- and gender-matched pairs (231 in all) of southern Chinese children ages 3 to 12 years, with and without cleft lip and palate. METHODS: Dental histories and radiographs were studied. From these, dental development was determined. RESULTS: Of 2946 tooth pairs in the children with cleft lip and palate, 252 (8.6%) were found to be asymmetric, significantly (p < .001) more than the 63 of 3179 (2.0%) tooth pairs observed in the children without cleft lip and palate. The teeth with the highest frequency of asymmetry were the maxillary lateral incisors (38.1%). Differences in dental development on the cleft versus noncleft sides of children with unilateral cleft lip and palate were significant for both maxillary and mandibular teeth (p < .001 and p = .039, respectively). The mean delay in tooth formation of the children with cleft lip and palate was 4.4 months relative to the children without cleft lip and palate. The majority of the teeth in the cleft lip and palate group were delayed by one developmental stage. This happened in 40.0% of the maxillary teeth and 30.1% of the mandibular teeth. The delay in tooth formation increased as the number of missing teeth increased in the children with cleft lip and palate, although not significantly. CONCLUSIONS: This group of southern Chinese children with cleft lip and palate demonstrated a higher prevalence of asymmetric and delayed dental development than did their counterparts who did not have cleft lip and palate.     

Different cleft conditions, facial appearance, and speech: relationship to psychological variables.

OBJECTIVE: The purpose of this investigation was to study the adjustment and learning characteristics of children with different types of clefts. The hypotheses were that there may be different relationships among cleft variables (speech and appearance) according to the cleft types. DESIGN: The study compared three cleft groups on behavior rating, anxiety scales, depression scales, and self-perception (analyses of variance) and examined the influence of facial and speech ratings on self-perception (multiple regression analyses). SETTING: All patients were treated at a university hospital cleft palate clinic. PATIENTS: Sixty-five children aged 8 years to 17 years were selected based on nonsyndromic cleft (unilateral cleft lip and palate [ULP], bilateral cleft lip and palate [BLP], and cleft palate only [CPO]) and no significant neurological condition or hearing loss. RESULTS: The findings indicated children with CPO showed greater problems with parent- and teacher-reported depression, anxiety, and learning related to speech than children with ULP or BLP. The later two groups showed fewer problems and a greater relationship of problem to facial appearance. The children with ULP self-reported lower levels of depression than the other two groups. CONCLUSIONS: Children with cleft show relatively good overall adjustment, but some problems appear related to speech and facial appearance. Subgroups may need to be studied separately.     

Type of oral cleft and mothers' perceptions of care, health status, and outcomes for preadolescent children.

OBJECTIVE: To evaluate the outcomes of care for children by type of oral cleft. DESIGN: Data were collected through structured telephone interviews during 2003 in Iowa with mothers of 2- to 12-year-old children with oral clefts. Interviews with mothers of children with clubfoot and statewide data on Iowa children were used for comparison. PARTICIPANTS: Participants included mothers of children in Iowa born between 1990 and 2000 with nonsyndromic oral clefts. Children were identified by the statewide Iowa Registry for Congenital and Inherited Disorders. MAIN OUTCOME MEASURES: Rating of cleft care, severity of condition, health status, esthetic outcome, speech, and school performance were evaluated by type of oral cleft. RESULTS: Children with cleft lip and palate were most likely to have their clefts rated as very severe. Children with palatal involvement were reported to have a lower health status and were almost twice as likely to be identified as having a special health care need compared with either children with cleft lip or children statewide. Children with cleft lip had more esthetic concerns; children with palatal involvement had the most speech concerns. CONCLUSIONS: Although mothers generally believed their children had received high-quality care, ratings of the children's current health status and outcomes of care varied significantly by type of cleft (cleft lip, cleft palate, and cleft lip and palate). Differences observed in this population-based study support the proposition that cleft type should be considered when examining outcomes of care for children with oral clefts.     

Conversational skills of children with cleft lip and palate: a replication and extension.

OBJECTIVE: To replicate and to extend a previous study examining the conversational skills of children with cleft lip and palate. PARTICIPANTS: Thirty-four children (33 to 44 months) participated: 17 children with cleft lip and palate and 17 noncleft children. METHODS: The children were observed during an interaction with caregivers in their homes. Samples of caregiver-child interactions were coded as assertive or responsive, for type of conversational act, and for discourse level categories. Profiles of conversational activity were determined for each child based on the coding. Correlations were performed to examine the relationship between assertiveness and speech variables (articulation and resonance) for the children with cleft lip and palate. RESULTS: Group comparisons revealed that the children with cleft lip and palate produced fewer assertive utterances, were less likely to respond adequately to comments by caregivers, and produced more topic maintaining and fewer topic extending utterances than did their noncleft peers during conversational interactions. Examination of individual child data indicated that 35% of the children with cleft lip and palate exhibited conversational profiles characterized by either low assertiveness or low responsiveness. Finally, a significant positive correlation was noted between conversational assertiveness and speech production skills. CONCLUSION: These findings suggested that the children with cleft lip and palate were less conversationally assertive than their noncleft peers. Further, there appeared to be a relationship between speech production skills and conversational skills, suggesting that poor speech may be impacting language performance for these children.     

Centre-based statistics of cleft lip with/without alveolus and palate as well as cleft palate only patients in Aden,Yemen

ObjectiveThe purpose of the study was to report the types and patterns of cleft lip with/without cleft alveolus and palate as well as cleft palate only as seen in Aden, Yemen.Design and settingRetrospective, centre-based study conducted at the Cleft Lip and Palate Centre, Aden University, Yemen.Material and methodsStatistical evaluation of the data from all cleft patients who were registered at or referred to this centre during the years 2005–2011.ResultsA total of 1110 cleft patients were seen during the period studied (2005–2011). Amongst these there were 183 (16.48%) with a cleft lip and 144 (12.98) with a cleft of lip and alveolus, 228 (20.54%) had a cleft palate, and 555 (50%) had a combination of cleft lip, alveolus, and palate. The clefts were found more often in males than in females (56.5% boys versus 43.5% girls). This difference was statistically significant (p ≤ 0.001). Statistically significant sex differences were also noted when evaluating the various cleft types. Isolated cleft palates were found most often in females. Among the cleft palate cases there were 102 (9.2%) with a cleft soft palate only. The ages of the patients were between one day and 40 years.Two hundred and one children (18%) had a positive family history of clefts. Among the risk factors considered in this study, consanguineous marriages among cousins were found most frequently (in 48% of the cases). In contrast to this, only 10% of the mothers had reported to have been taking medication directly prior to or during the first trimester of their pregnancy. On average the mothers were neither very young nor very old.ConclusionThe prevalence rate of orofacial cleft types among this Yemeni sample was similar to prevalence rates previously reported in white Caucasians. The present study did neither find many cases with medication before, nor during, pregnancy; there were few young or very old mothers; and the incidence of positive family histories was similar to those found in other studies on clefts. However, consanguineous marriages were encountered quite often.     

Retrognathic maxilla in individuals born with oral clefts is due to intrinsic factors and not only due to early surgical treatment

ObjectivesTo determine if the skeletal form of individuals born with oral clefts was associated with maxillary position.Materials and MethodsLateral cephalometric radiographs of 90 individuals 8 to 12 years old born with or without cleft lip and palate paired by age and sex were used. Skull base length, cranial base angle, cranial deflection angle, and maxillary skeletal length and position were studied. Also, mandibular skeletal length and position, lower anterior facial height, and dental position were defined. Individuals were divided into three groups: 30 individuals born with cleft lip and palate with Class III malocclusion (UCLP Class III), 30 individuals born with cleft lip and palate with Class I malocclusion (UCLP Class I), and 30 individuals born without cleft lip and palate with Class III malocclusion (non-cleft Class III).ResultsWhen comparing the UCLP Class III group with the UCLP Class I group, there were differences in maxillary position (P < .001) and mandibular position (P = .004) found. No differences were found when comparing the UCLP Class III group with the non-cleft Class III group.ConclusionsThere are intrinsic factors that affect craniofacial morphology of individuals born with cleft lip and palate.     

Intravelar veloplasty in cleft lip, alveolus and palate and outcome of speech and language acquisition: a prospective study

Background: Speech and language acquisition are major, important criteria in the treatment outcomes of cleft lip and palate patients. A generally accepted and definitive treatment protocol regarding surgical techniques and the time schedule does not yet exist. In the world literature, there are reports of velo-pharyngeal insufficiency rates between 7 and 30%.Purpose: In a prospective study, all children aged months with cleft lip, alveolus and palate, or cleft palate only, underwent an intravelar veloplasty. Follow-up monitoring consisted of frequent clinical linguistic checks and supervision of language development without a planned intention of articulation therapy before the age of about 5 years.Results: Three hundred and ninety-seven children with non-syndromic clefts were included in this study, the youngest being 8-year old. Sixty children (15%) showed deviations in language and speech acquisition. From these, 56 (14%) had received articulation therapy after the 5th birthday. From these 56 children, 45 had overcome their problems with speech therapy alone whereas 11 (3%) needed a velo-pharyngeoplasty.Discussion: Although these results are much better than those reported in other cohorts, some children still have velo-pharyngeal incompetence for no apparent reason. One possible explanation might be surgical, since on occasions, the intravelar muscle bundle is divided into two parts and the palato-pharyngeal part runs isolated more laterally and can be missed during reconstruction and retropositioning.     

Habilitation of patients with clefts: parent and child ratings of satisfaction with appearance and speech.

This study examined ratings regarding satisfaction with facial appearance and speech performance from 495 parent-child pairs. Data were obtained from school-aged children (5-18 years old) and their parents using standardized independent interviews. Results revealed that 54 percent of the children with cleft lip (CL) or cleft lip and palate (CLP) were very pleased with their appearance, and 62 percent of the cleft palate or CLP subjects were very pleased with speech. Low, but statistically significant correlations exist between the female subjects and their parents in satisfaction with appearance. Although older cleft lip and/or palate patients reported increased satisfaction with speech, no age differences in patient satisfaction with appearance were observed in subjects with CL/CLP. Parents of females expressed more concern about their daughters' appearance than parents of males, while parents of males were more concerned about speech. Implications for craniofacial habilitation teams and research suggestions are discussed.     

A cross-sectional study of speech in 10-year-old children with cleft palate: results and issues of rater reliability.

OBJECTIVES: To describe speech based on perceptual evaluation in a group of 10-year-old children with cleft palate. A secondary aim was to investigate the reliability of speech-language pathologists' perceptual assessment of cleft palate speech. DESIGN: Retrospective cross-sectional study in children with cleft palate. External raters made assessments from randomized speech recordings. SUBJECTS: Thirty-eight children with unilateral cleft lip and palate (UCLP) or cleft palate only (CPO) and 10 children in a comparison group. MAIN OUTCOME MEASURES: Ratings of hypernasality, hyponasality, audible nasal air leakage, weak pressure consonants, and articulation. Exact agreement and weighted kappa values were used for reliability. RESULTS: Hypernasality was found in 25% of children with a cleft of the soft palate (CSP), 33% of children with a cleft of the hard and soft palate (CHSP), and 67% of children with a UCLP. Similar results were found for audible nasal air leakage. Articulation errors were found in 6% of the CHSP group and 25% of the UCLP group, whereas no child in the CSP group had articulation errors. The reliability was moderate to good for different variables, with lowest values for hypernasality. CONCLUSIONS: Speech results in this series seem less satisfactory than those reported in other published international studies, but it is difficult to draw any certain conclusions about speech results because of large methodological differences. Further developments to ensure high reliability of perceptual ratings of speech are called for.     

Parent and child ratings of satisfaction with speech and facial appearance in Flemish pre-pubescent boys and girls with unilateral cleft lip and palate

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.     

Speech Outcome After Cranial-Based Pharyngeal Flap in Children Born With Total Cleft, Cleft Palate, or Primary Velopharyngeal Insufficiency

PURPOSE: The aim of this study was to compare the effect of a cranial-based pharyngeal flap on the speech of children born with a unilateral cleft lip and palate (UCLP), bilateral cleft lip and palate (BCLP), cleft palate (CP), or primary velopharyngeal insufficiency (VPI) without cleft. PATIENTS AND METHODS: A total of 234 children born with clefts and 22 children born with primary VPI were evaluated. Children with associated abnormalities were excluded from this study. The Borel-Maisonny classification system was used to evaluate the velar insufficiency. The cranial-based pharyngeal flap was performed using the Sanvenero-Rosselli technique. RESULTS: Between 1984 and 2001, 74 children underwent pharyngeal flap for VPI. The mean follow-up period was 7 years. Borel-Maisonny scores after pharyngeal flap surgery were as follows: children with UCLP (n = 22), 59.1% type 1, 36.4% type 1/2, and 4.5% type 2; children with BCLP (n = 18), 44.4% type 1, 27.8% type 1/2, 16.7% type 2, and 11.1% type 2/3; children with CP (n = 17), 64.7% type 1, 23.5% type 1/2, and 11.8% type 2; children with primary VPI (n = 17), 29.4% type 1, 29.4% type 1/2, 29.4% type 2/3, and 11.8% type 3. There were significant differences in outcome among the 4 groups (P = .029; Fisher exact test). CONCLUSIONS: The positive effect on speech of a cranial-based pharyngeal flap is greater in children born with a UCLP or CP than in those born with a BCLP. In children born with primary VPI, this operation has only a slightly positive effect on speech that shows compensatory misarticulations; in such cases, alternative surgical choices or secondary procedures may be indicated. This information should be clearly conveyed to the parents in presurgical consultation so that they know what to expect from the procedure and postoperative adjuvant therapy.     

2011—2015年广东省唇腭裂流行状况分析

目的了解2011—2015年广东省唇腭裂流行状况。 方法以2011—2015年在广东省出生缺陷监测体系内58家医院分娩的围产儿（孕28周至出生后7天）及孕产妇为研究对象,描述性分析广东省唇腭裂分布及类型特征,使用卡方检验、有序分类回归,构成比进行统计学分析。 结果符合纳入标准的围产儿1 203 800例,检出唇腭裂1664例,检出率为13.83/万;广东省内揭阳市检出率最高为21.30/万,湛江市检出率最低为7.78/万。2011—2015年逐年检出率分别为15.81/万、14.00/万、9.02/万、15.81/万、11.00/万,各年份间无明显趋势性;总唇腭裂检出率在4月最高和9月最低,差异无统计学意义（χ²= 128.34,P= 0.403）。孕产妇年龄分层中,>35岁年龄组和<20岁年龄组检出率较高,分别为23.86/万和17.73/万;男性围产儿检出率为16.45/万,女性围产儿的检出率为12.40/万,性别间总唇腭裂的检出率差异有统计学意义（χ²= 32.74,P<0.0001）,唇腭裂类型在不同围产儿性别间的分布其差异有统计学意义（χ²= 52.78,P= 0.0012）。唇裂、腭裂、唇裂合并腭裂的构成比分别为27.94%、25.54%、46.51%。 结论加强唇腭裂区域防控措施,进一步研究唇腭裂性别差异的影响因素,普及优生优育及出生缺陷防控相关健康教育以减少出生缺陷的发生。     

Type of reading disability related to cleft type and neuropsychological patterns

Two groups of cleft palate subtypes (cleft lip and palate and cleft palate only) with reading disability, matched for intelligence, age, sex, and reading level, were compared on reading and neuropsychological test variables. Subjects included 14 males and 10 females of each cleft type ranging in age from 8 to 13. Results showed a significant difference between groups on most language measures and differences in reading comprehension and type of reading errors. Results suggest that children with cleft palate only constitute a language-disorder group with more severe reading disabilities. Children with cleft lip and palate are more likely to have verbal expressive deficits and milder reading problems, possibly related to peripheral speech mechanisms.     

Predictors of speech outcomes in children with Pierre Robin sequence

Backgound

Pierre Robin sequence (PRS) has worse speech outcomes than isolated cleft palate. We aimed to search for possible associations of phonological outcomes with PRS status (isolated vs syndromic), clinical severity, soft palate muscles deficiency, or surgical procedure.

A preliminary study of nasal airway patency and its potential effect on speech performance.

The relationship between nasal airway size and articulatory performance was studied in a group of cleft palate patients. Articulation analysis revealed that children with bilateral cleft lip and palate were nearly twice as likely to manifest compensatory articulations as children with unilateral cleft lip and palate or with cleft palate only. When subjects were grouped according to speech performance, aerodynamic assessment indicated that children with compensatory articulations had significantly larger nasal cross-sectional areas than children without compensatory articulations. The findings suggest that children with comparatively large nasal airways may be at increased risk for developing abnormal speech patterns. If these findings are confirmed by further research, such children may be candidates for relatively early palate repair.     

Evaluation of Speech Disorders in Children with Cleft Lip and Palate

AbstractBackground: We investigated the reliability of nasopharyngoscopy and auditory perception, two common clinical methods employed in examining the speech pathology of children with cleft lip and palate.Patients and Methods: Nasopharyngoscopy was performed to assess velopharyngeal closure function and nasopharyngeal morphology. The auditory examination evaluated nasality and other speech disorders such as articulatory tension, shift in articulation, and sigmatism. The study was based on the audio and video recordings of twelve patients (9.5 ± 0.5 years) after surgical rehabilitation of their cleft lip and palate. The speech sample consisted of reading a standardized sequence of words. The recordings were analyzed by several examiners with different experience in the evaluation procedure at two time-points. Inter-rater and intra-rater reliability was determined by Cohen’s Kappa coefficient and by estimation of the 95% intervals of confidence.Results: Analysis of the visual and auditory-perceptual findings by the experienced raters was highly reliable. However, the less experienced raters’ results showed greater variability, as did morphological evaluation in both groups. Comparison between visual and auditory evaluations concerning hypernasality resulted in little correlation.Conclusions: These two subjective methods of evaluation are recommended because of their high reliability with experienced raters. Our results suggest that “experience” is an essential criterion in determining the structural quality of the morphological and functional assessment of typical speech pathologies in children with cleft lip and palate. In addition, the data demonstrate the necessity of special multidisciplinary rehabilitation teams in so-called cleft centers.     

Type of facial cleft, associated congenital malformations, and parents' ratings of school and conduct problems

Parents' ratings of conduct and school problems were evaluated in 79 subjects with palatal clefts. Subjects were divided into four groups consisting of: (1) unilateral complete clefts of the lip and palate (UC Group); (2) unilateral complete clefts of the lip and palate with associated congenital malformations (UC-C Group); (3) cleft palate only (CPO Group); and (4) cleft palate only with associated congenital malformations (CPO-C Group). Comparisons between groups suggested that subjects with associated congenital malformations, whether or not they had UC or CPO, tended to have more serious school and conduct problems when compared to children with clefts and no associated congenital malformations. The association of congenital malformations with a cleft lip and palate increased the risk of reported conduct problems at home and behavioral and learning problems at school.