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.     

A descriptive epidemiology study of cleft lip and palate in Northern Finland

Objective. The aim of this study was to determine the incidence of cleft lip and/or cleft palate in a population uniquely from Northern Finland. Materials and methods. The records of a total of 214 cleft patients treated between 1998–2011 at the Oulu Cleft Lip and Palate Center at the University of Oulu were assessed on a retrospective basis. Data regarding cleft type, sex and side of cleft was collected and analyzed. Family history of clefting was investigated. Results. Cleft palate (68.7%) was most frequently found, followed by cleft lip and palate (18.7%) and cleft lip with or without alveolus (12.6%). Cleft palate occurred more frequently in females (63.3%) and cleft lip and palate was more frequently found in males (62.5%). The left side was more frequently affected in both male and female patients. Left-sided clefts were observed in 82% of patients compared to right-sided clefts in 18%. A family history of clefting was detected in 20.1% of patients. Conclusion. The incidence of clefts in Northern Finland is higher than the corresponding incidence in other European countries. Cleft palate was the most frequent cleft type and it was more frequent in females. In males, cleft lip and palate was more frequent. The left side was more frequently affected in both genders. One fifth of the patients had a family history of clefts.     

河南省部分地区唇腭裂患者术后继发畸形调查分析

目的：了解唇裂、腭裂一期手术后继发畸形的情况。并探讨其影响因素及有效的解决方法。方法：采用普查法对河南省部分地区人群进行随访,对唇腭裂患者进行术区照相,根据照片记录手术类型、术后继发畸形情况,将结果进行统计学分析。结果：共随访唇腭裂术后患者2046例。单侧唇裂患者1514例,术后继发畸形1268例,,双侧唇裂患者488例,术后继发畸形466例,腭裂患者446例,术后继发硬腭穿孔及软腭复裂26例。结论：适当选择唇腭裂手术类型,对唇腭裂术后患者进行定期随诊及普查,及时发现、治疗术后继发畸形,对提高唇腭裂手术的治疗效果具有极为重要的意义。     

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 population-based evaluation of antenatal diagnosis of orofacial clefts

OBJECTIVES: To evaluate the changes in prevalence and antenatal detection of cleft lip with or without cleft palate and isolated cleft palate and to describe the association between anomalies and rates of antenatal diagnosis in Nova Scotia from 1992 to 2002. DESIGN: This population-based cohort study employed the Nova Scotia Atlee Perinatal Database, the Fetal Anomaly Database, and IWK Cleft Palate Database in Halifax, Nova Scotia, Canada. OUTCOME MEASURES: Cleft type, mode of diagnosis, and associated abnormalities of orofacial clefts for liveborn infants, stillbirths, and second trimester terminations of pregnancy between 1992 and 2002 were determined. RESULTS: There were 225 fetuses identified as having orofacial clefts. The overall prevalence of clefts was 2.1 in 1000 live births, and this prevalence did not change with time. The overall antenatal detection of cleft lip with or without cleft palate was 23%; however, there was improvement in detection of cleft lip with or without cleft palate from the years 1992 to 1996 (14%) to the years 1997 to 2002 (30%, p = .02). No isolated cleft palates were detected antenatally. Associated structural anomalies were seen in 34.2% of cases with orofacial clefts, and chromosomal abnormalities were associated with 9.8%. CONCLUSIONS: The prevalence of orofacial clefts in Nova Scotia has not changed from 1992 to 2002. The proportion of antenatally diagnosed cleft lip with or without cleft palate in Nova Scotia is consistent with rates reported in the literature and has increased from 1992 to 2002.     

Sucking performance of babies with cleft conditions.

OBJECTIVE: To describe the sucking performance of bottle-fed babies with cleft conditions. PARTICIPANTS: Forty 2-week-old-babies with cleft lip (CL; n = 8), cleft palate (CP; n = 22), and cleft lip and palate (CLP; n = 10) were examined. METHODS: Suction, compression, and other sucking parameters were measured during bottle-feeding and compared to determine if they varied with cleft condition or feeding ability. RESULTS: All babies with CL and one with CLP demonstrated suction. Thirteen of 22 babies with CP demonstrated suction but only three maintained regular pressure changes over time. Between-group differences in the amplitude of suction and compression were associated with cleft condition. Cleft lip participants demonstrated the greatest amplitude of suction followed by those with CP and CLP. Cleft lip and CP participants generated similar amplitudes of compression. This was greater than their counterparts with CLP. Good feeders (n = 15) generated high levels of suction, while satisfactory (n = 15) and poor feeders (n = 10) did not generate any during bottle-feeding. CONCLUSION: Between-group differences in intra-oral pressures were confirmed when babies were examined by cleft condition. Babies with smaller clefts (i.e., CL or minor soft palate clefts) were more likely to generate normal levels of suction and compression compared to their counterparts with larger clefts. Since good feeders were more likely to have smaller clefts it was not surprising that they demonstrated higher suction pressures than babies with satisfactory or poor feeding ability. Compression values were not significantly different across the feeding ability groups. These data may inform feeding management strategies.     

The incidence of cleft lip and palate deformities in the south-east of Scotland (1971-1990).

This retrospective study reports the incidence of infants born with the cleft lip and palate anomaly within the Edinburgh Cleft Units catchment area, between 1 January, 1971, and 31 December, 1990. The importance of accurate data collection for local, regional, and national data bases is discussed with reference to the recent CSAG report on cleft lip and palate services in the UK. Five-hundred-and-two cleft lip and palate patients were identified (291 males, 211 females). The incidence is reported as 1.4 per 1000 live births (1 in 711). Twenty-five per cent of clefts affected the primary palate, 45 per cent affected the secondary palate, and the remaining 30 per cent were clefts of both the primary and secondary palate. Overall, a higher percentage of males were affected (58 per cent males to 42 per cent females). Clefts of the secondary palate, however, were more common in females (56 per cent females to 44 per cent males). Data presented in this study is similar to that previously reported from UK centres. It is suggested the accuracy of the UK cleft lip and palate data collection needs to be improved. Prospective data collection in a standardized format carried out on a national basis has to be a priority as recommended by the CSAG report.     

Major hematological diseases associated with cleft lip and palate.

OBJECTIVE: Cleft lip and palate is among the most common congenital anomalies. Its association with major blood disorders has rarely been reported. The purpose of this study was to report two patients who had major blood diseases associated with cleft lip and palate. PATIENTS AND RESULTS: From June 1995 to December 1997, there were 2700 patients with cleft lip, cleft palate, or both who received treatment at Chang Gung Memorial Hospital. Two of them were found to have major hematological disorders. In both cases, the disorder was detected by preoperative blood cell counts and white cell differentiation. Case 1 was a 21-year-old woman patient with repaired right cleft lip. She was admitted for alveolar bone grafting and closure of oronasal fistula. Abnormal presentation of blast cells was found, and subsequent bone marrow study confirmed acute lymphocytic leukemia. Case 2 was a 26-year-old man with left secondary cleft lip nasal deformity scheduled to receive staged reconstructive operations. An elevated platelet count was found and subsequently confirmed to represent essential thrombocytosis. In both cases, reconstructive operations for the cleft-related deformities were performed. CONCLUSIONS: Association of major hematological disorders and cleft lip, palate, or both is rare and is reported herein.     

Psychological study of cleft palate children with or without cleft lip by Kinetic Family Drawing

To clarify how cleft palate children with or without cleft lip perceive their family, we applied the Kinetic Family Drawing (KFD) technique to 60 cleft palate children (with or without cleft lip). The children were aged 7-9 years (in the 1-3 grade of elementary school) and attended the Department of Oral Surgery, Nagoya University Hospital, between 1990 and 1997. Controls consisted of 100 normal elementary school children of the same age. Major findings were: (1) Compared with normal children, cleft lip and palate boys and cleft palate only girls drew their self-images significantly more often in region D (lower right) and in region A (upper left), respectively. (2) Cleft palate children with or without cleft lip felt anxiety and fear toward their family, rarely viewing their homes as a restful place; this suggested insecurity. Uniquely dynamic relations were also suggested in their homes. (3) Cleft palate children with or without cleft lip often perceived their father or mother as persons easily acceptable in society. Rather remote psychological distance between parents and cleft palate children with or without cleft lip was suggested.     

基于圆形分布法的广东省唇腭裂患儿受孕月份分析

目的探讨广东省唇腭裂患儿受孕月份的分布规律。 方法以2016—2017年在广东省范围内有产科资质的医疗机构内分娩的围产儿（孕28周至出生后7 d）及产妇为研究对象,将唇腭裂患儿受孕统计年度看成1个圆周,用圆形分布法对其中的先天性唇腭裂患儿的受孕时间进行聚集性分析。 结果2016—2017年广东省共监测围产儿3 748 435例,检出唇腭裂患儿5591例,其中男性患儿3137例、女性患儿2309例、性别不明或两性畸形患儿145例,排除性别不明患儿共包括唇裂1368例、腭裂913例、唇裂合并腭裂3165例。唇腭裂患儿的受孕高峰有时间聚集性（平均角 = 67.33,P<0.001）,唇腭裂患儿的受孕高峰在3月上旬。其中单纯唇裂和唇裂合并腭裂的受孕高峰时间分别是2月下旬和3月中旬,为春季;单纯腭裂的受孕高峰时间差异无统计学意义。女性唇腭裂患儿的受孕时间存在高峰,为3月份,男性患儿的受孕时间差异无统计学意义。 结论广东省唇腭裂患儿的受孕高峰有时间聚集性,单纯唇裂和唇裂合并腭裂与单纯腭裂可能有不同病因构成,女性患儿的发病更受到季节性影响,春季应加强预防唇腭裂危险因素的暴露。     

Left-sided predominance of hypodontia irrespective of cleft sidedness in a French population

Objective : Individuals with oral clefts exhibit considerably more dental anomalies than do individuals without clefts. Our aim was to evaluate the prevalence of tooth agenesis in a sample composed of 124 children (81 boys and 43 girls, mean age 12.5?years) with clefts registered with the Cleft Palate Center in Strasbourg (France). Design : Cleft types and dental agenesis were assessed by clinical and radiographic examination. Cleft types were divided into four groups according to the clinical extent of the cleft (cleft lip only [CL, 12.9%], cleft lip and alveolus [CLA, 4%], cleft lip and palate [CLP, 49.2%], and cleft palate only [CP, 33.9%]). Results : Of the subjects 63% had evidence of hypodontia: maxillary lateral incisors (54%) and upper and lower premolars (32%) were the most common missing teeth. The number of dental ageneses associated with CP (54%) and CLP (79%) was significantly higher than that with CL (33%). All these anomalies were found in proportionately higher frequencies as the severity of the cleft increased, and we found left side predominance for hypodontia (p < .01) irrespective of cleft sidedness. Conclusions : Both right-sided and left-sided clefts were more frequently correlated with left-sided dental agenesis. This left-sided prevalence suggests that common signaling malfunctions might be involved, both in dental development anomalies and cleft genesis.     

Research Reports

Abstract

This retrospective study reports the incidence of infants born with the cleft lip and palate anomaly within the Edinburgh Cleft Units catchment area, between 1 January, 1971, and 31 December, 1990. The importance of accurate data collection for local, regional, and national data bases is discussed with reference to the recent CSAG report on cleft lip and palate services in the UK.

Five-hundred-and-two cleft lip and palate patients were identified (291 males, 211 females). The incidence is reported as 1.4 per 1000 live births (1 in 711). Twenty-five per cent of clefts affected the primary palate, 45 per cent affected the secondary palate, and the remaining 30 per cent were clefts of both the primary and secondary palate. Overall, a higher percentage of males were affected (58 per cent males to 42 per cent females). Clefts of the secondary palate, however, were more common in females (56 per cent females to 44 per cent males).

Data presented in this study is similar to that previously reported from UK centers. It is suggested the accuracy of the UK cleft lip and palate date collection needs to be improved. Prospective data collection in a standardized format carried out on a national basis has to be a priority as recommended by the CSAG report.     

Incidence of cleft lip and palate in the northeast of Mexico: a 10-year study

In Latin American countries, studies on the incidence of cleft lip and palate are not widely available. A 10-year retrospective study was carried out to determine the incidence of cleft lip and palate at the Autonomous University of Nuevo Leon's University Hospital. The study included data from patients who attended the plastic surgery outpatient clinic from January 1990 to December 1999. The author reviewed 10,843 files from which 376 patients were selected to identify the following variables: time of the year in which the first consultation took place, gender, birthplace, type of cleft, age of both parents, and medications taken during pregnancy. The highest incidence was found in patients aged 1 to 6 months. Ninety-four patients had a primary palate cleft; 76 had a secondary palate cleft; and 206 had primary and secondary palate clefts. The gender distribution of the 206 patients with primary and secondary clefts was 127 boys and 79 girls. The mean parent age was 29.5 years (father) and 25.7 years (mother). The incidence of cleft lip and palate in the cited hospital was 1.1:1000 births. Clefts of the left side occurred more often than of the right. Boys were affected more commonly than girls.     

Epidemiology of Hoffmeister's "genetically determined predisposition to disturbed development of the dentition" in patients with cleft lip and palate.

OBJECTIVE: Type and prevalence rates of the symptoms of Hoffmeister's "genetically determined predisposition to disturbed development of the dentition" were studied in patients with clefts. PATIENTS: Data of 263 patients with nonsyndromic clefts of lip (alveolus and palate) or isolated cleft palates were examined in a retrospective study. SETTING: The clefts were classified as cleft lip or cleft lip and alveolus, cleft palate, unilateral cleft lip and palate, and bilateral cleft lip and palate. All patients were scrutinized for 28 individual symptoms. Prevalences of the individual symptoms were statistically evaluated regarding cleft type and gender by using the chi-square test and were also compared with findings in patients without clefts. RESULTS: In 97.7% of the patients with clefts, at least one symptom was found. Microdontia of individual teeth, hypodontia, and hyperodontia were the symptoms most frequently recorded. Comparison of the different cleft types revealed differences regarding the prevalences of supernumerary lateral incisors (p = .051), infraposition of deciduous molars (p < .001), and atypical tooth bud position (p = .030). Comparison of the prevalences of 10 symptoms recorded in the patients with clefts with the prevalences recorded in patients without clefts showed nine symptoms were found much more frequently in the population with clefts. CONCLUSION: These findings support the hypothesis that clefting is part of a complex malformation associated with other dental anomalies resulting from disturbed development of the dentition. Patients with clefts are also likely to present other deficiencies of dental development and tooth eruption in both dentitions, even in regions not affected by the cleft.     

Questionnaire survey of attitudes and concerns of patients with cleft lip and palate and their parents

The self-perceived problems and concerns of patients with complete unilateral cleft lip and palate and their parents were investigated. Thirty-two patients ranging in age from 16 to 25 years (mean = 19.9 years) and their parents were sent questionnaires by mail. Twenty-eight patients and 30 parents completed the questionnaires. The results suggest that the patients were satisfied with the treatment they had received. They believed that the efforts of the members of the cleft palate team had been successful, and they were generally satisfied with their overall facial appearance and speech. There were, however, specific aspects with which they were less satisfied; their nose, lip, profile, speech, and teeth. They did not feel significantly socially or emotionally handicapped, although they noted that they had been teased and that they found relations with the opposite sex difficult. Their parents were also generally satisfied with all aspects of treatment and their child's appearance and speech. They identified those areas that their child was most concerned with, but did not fully appreciate how satisfied their child was with his or her facial features. The parents felt that their children were socially and emotionally affected by their cleft and felt their school results had also been affected.     

Oral health-related quality of life, and satisfaction with treatment and treatment outcomes of adolescents/young adults with cleft lip/palate: an exploration

The objectives of this correlational study were to explore whether the oral health-related quality of life (ohrqol) of adolescents/young adults with cleft lip/palate (CL/P) relates to their own and their parents’ satisfaction with treatment and treatment outcomes. Data were collected using mailed surveys from 30 parents and 27 patients who completed CL/P treatment. Patients’ ohrqol was assessed with the Michigan Oral Health-Related Quality of Life (MOHRQoL) Scale, treatment satisfaction with Kiyak's Post-Surgical Patient Satisfaction Questionnaire, and satisfaction with treatment outcomes with Noor and Musa's Cleft Evaluation Profile. Overall, patients reported a positive ohrqol. Their satisfaction scores ranged from low to high. Patient and parent treatment satisfaction was related, while their outcome satisfaction did not correlate. Patients’ MOHRQoL scores correlated with parent and patient treatment satisfaction and parents’ outcome satisfaction. Patients’ MOHRQoL scores did not correlate with patients’ outcome satisfaction. The patients’ level of discomfort was strongly correlated with patients’ and parents’ treatment satisfaction and parents’ outcome satisfaction. In conclusion, regardless of outcome satisfaction, young patients with CL/P report a quite positive ohrqol after treatment completion. There is a clear relationship between the ohrqol of these patients with CL/P and their own as well as their parents’ treatment satisfaction assessments.     

Parent reports of the psychosocial functioning of children with cleft lip and/or palate.

OBJECTIVES: (1) to determine the opinion of parents regarding the psychosocial functioning of their child with cleft lip and/or palate (CLP); (2) to identify predictors of psychosocial functioning; and (3) to determine the level of agreement between children with CLP and their parents. Participants: One hundred twenty-nine parents of children with CLP and 96 parents of children without CLP participated in this cross-sectional study. OUTCOME MEASURES: Parental opinion of the child's self-esteem, anxiety, happiness, and problems caused by facial appearance were assessed using visual analogue scales. Parents completed the Child Behavior Checklist and were interviewed. RESULTS: Children with CLP were more anxious (p < 0.05), less happy with their appearance (p < 0.001), and in general (p < 0.05) had lower self-esteem (p < 0.05) and greater behavioral problems (p < 0.001) compared with non-CLP children. Parents reported that their child with CLP was teased more often (p < 0.001) and was less satisfied with his/her speech (p < 0.01) compared with reports of parents in the control group. A number of factors affected parents' ratings of their child's psychosocial functioning (presence of CLP, appearance happiness, previous history of CLP, and visibility of scar). Children who had been teased were more anxious (p < or = 0.01), less happy with their appearance (p < 0.001) and had greater behavioral problems (p < 0.001). CONCLUSIONS: Parents of children with CLP reported various psychosocial problems among their children. Parents considered children who had been teased to have greater psychosocial problems.