Provision of general dental care for children with cleft lip and palate--parental attitudes and experiences

OBJECTIVE: To evaluate the attitudes of parents of 4-8 year-old children with cleft lip and palate (CLP) towards the provision of paediatric dental care and to assess their experience of treatment within the General Dental Services. DESIGN: Postal questionnaire distributed to all parents of 4-8 year-old children on the Birmingham CLP database. RESULTS: The response rate was 77%. Ninety-nine (91%) children were registered with a dentist. Seventy-five (69%) had previously received preventive advice and 32 (29%) had experienced restorative intervention. The majority of parents (64%) expressed a wish for a dental check-up to be provided at the designated Cleft Centre, with 42 (39%) requesting preventive advice. Fifty-eight (67%) of the parents who requested a dental check-up were agreeable for treatment to be provided in the primary sector. CONCLUSION: The survey indicates there is parental support for paediatric dental assessment at cleft clinics with subsequent arrangement of treatment in the primary sector. The inclusion of paediatric dental support within the multidisciplinary cleft team should be considered as Regional Cleft Centres are established     

Association of nasomaxillary asymmetry in children with unilateral cleft lip and palate and their parents.

OBJECTIVE: It has been suggested previously that increased width of midfacial structures is associated with the development of palatal clefting. The aim of this study was to evaluate the association of transverse craniofacial asymmetry between children with unilateral cleft lip and palate (UCLP) and their parents. Specifically, we hypothesized that parental transverse craniofacial asymmetry is a risk factor associated with the development of asymmetry in children with UCLP. DESIGN: Retrospective cross-sectional investigation including affected children and their noncleft parents. PATIENTS, PARTICIPANTS: A total of 64 children-parent sets of data (32 child-biological mother + 32 child-biological father) were included. Subject records included posteroanterior cephalometric radiographs obtained from 29 Costa Rican families with UCLP. MAIN OUTCOME MEASURES: The side of parental nasal asymmetry was significantly associated with the side of cleft in their children. For the majority of parents with children suffering from a left cleft, nasal width was larger on the left, compared with the right side. Similarly, in the majority of parents with children suffering from a right cleft, nasal width was larger on the right, compared with the left side. CONCLUSION: The results suggest that unilaterally increased nasomaxillary width in parents may play a key role in the development of ipsilateral palatal clefting in their offspring, therefore underscoring the importance of craniofacial form as a genetic etiologic factor in the genesis of clefting. Better understanding of the role of craniofacial form in cleft development will ultimately allow for the assessment of risk for cleft lip and palate.     

Parental craniofacial morphology in orofacial clefting

The parental craniofacial morphology in orofacial clefting (OFC) has been shown to differ from that of the non-cleft population when evaluated using conventional cephalometric analyses comprising a variety of linear, angular, and area measurements. In spite of this, the shape of the parental craniofacial complex is of greater importance in the search for the morphogenes involved in OFC. This retrospective case-control study employed three morphometric techniques [discriminant analysis of the principal components of shape (PCS), Euclidean distance matrix analysis (EDMA), and thin-plate spline analysis (TPS)] to localize the craniofacial skeletal shape differences between (a) the parents of children with OFC and a comparison group, (b) the parents of children with cleft lip and palate [CL(P)] and cleft palate (CP), and (c) the male and female parents of children with OFC. The postero-anterior (PA) cephalograms of 92 parents of children with non-syndromic OFC and 43 comparison group volunteers were scanned and digitized. The configurations of 24 reproducible landmarks were optimally superimposed using Procrustes algorithms to allow shape data to be derived using PCS, EDMA, and TPS. The parental craniofacial shape statistically significantly differed from that of the comparison group using PCS (P < 0.001) and EDMA (P = 0.001). However PCS, EDMA, and TPS differed in their localization of the shape differences, explainable by the different mathematical methods used by the individual techniques. Interestingly, the parental craniofacial shapes in CL(P) and CP were morphologically similar when tested using PCS (P = 0.03) and EDMA (P = 0.027). However, there was no shape-related sexual dimorphism in parental craniofacial morphology in OFC when tested using PCS (P = 0.35) and EDMA (P = 0.525). Thus, the parental craniofacial shape in OFC differs from the non-cleft population, the parental craniofacial shape does not differ between CL(P) and CP and there is no sexual dimorphism in the parental craniofacial morphology in OFC, as viewed on PA cephalograms.     

A local perspective on the initial management of children with cleft lip and palate by primary care physicians.

OBJECTIVE: To evaluate the frequency and referral patterns, need for continuing education, and information given to parents of children with cleft lip and palate by local primary care physicians. STUDY DESIGN: A survey was sent to primary care physicians from the pediatrics, family practice, and internal medicine/pediatrics specialties in six surrounding counties of a regional craniofacial center located within northeastern Ohio with a population base of 1.5 million people. Particular aspects of care evaluated included protocols of care, information for parents, referral patterns, conferences attended, and literature read. RESULTS: Nearly 30% of physicians had standard protocols when children are born with clefts. Eighty-seven percent of physicians referred children within 2 weeks of diagnosis, yet only 48% gave referrals to a multidisciplinary team. Physicians providing team referrals were nearly three times as likely to attend related conferences and approximately four times as likely to have a set plan at the time of diagnosis of the cleft. Forty-nine percent of physicians gave oral information, 35% gave oral and written information, and 10% did not give parents information. Ninety-four percent of physicians gave parents the option to call with concerns. Pediatricians were approximately four times as likely to attend cleft conferences and desire continuing education updates. However, family practitioners were over three times as likely to give proper referrals, compared with pediatricians. CONCLUSION: There is a necessity to educate local primary care physicians who manage children with cleft lip and palate regarding cleft information for parents, referral guidelines, the cleft team concept, and the role of primary physicians.     

Ethics for orthodontists.

When treating children, important ethical issues may arise relating to the best interests of the child and decision making for minors. The case of a child with a cleft lip and palate whose parents failed to bring her in for medically indicated orthodontic care is presented. Ethical features of the case are discussed, including the need to benefit the patient, avoid harm, and respect the preferences of the parents. Ethical codes of the American Dental Association and American Medical Association are referenced. Ethical dilemmas include the conflict between the orthodontist's obligation to the child and the need to respect parental autonomy. Parental autonomy is respected up until the point at which significant harm to a child may result. The orthodontist's primary ethical responsibility is to the child, not the parents. The orthodontist providing medically indicated care should involve the craniofacial team or hospital social worker when parental decision making is in question.     

Beyond easy answers: prenatal diagnosis and counseling during pregnancy.

OBJECTIVE: The advancing sophistication and availability of prenatal diagnostic technologies, such as transvaginal ultrasound, chorionic villus sampling, amniocentesis, and alpha feto-protein testing, have increased the medical capacity to detect genetic and congenital conditions during pregnancy. This paper raises many social and ethical questions about how families, craniofacial teams, and society respond when a prenatal diagnosis is made and considers the ethical and social issues around counseling, managing information, and making decisions. DESIGN: Ethical and sociological analysis. SETTING: Implications examined on the societal, health professional, and family level. RESULTS: Families and health professionals often manage ambiguity, uncertainty, and complex decision making in facing a prenatal diagnosis. Embedded in parental and clinical decisions are values about children with birth defects. Families are making decisions about whether to bear or abort an affected fetus on the basis of their perceptions of the impairment and on their expectation of the burden involved for the family and the child. On a broader, societal level, pressures to conform and minimize human differences are apparent in biomedical interventions, the Human Genome Project, advertising and media images, and social pressures to normalize disabilities. How society deals with prenatal diagnosis will impact upon social values; moral, legal, and ethical perspectives; and on health policy. CONCLUSION: Prenatal diagnostic technologies raise complex ethical, family, policy, and legal issues that have broad implications for the lives of children born with special health care needs, including children with cleft lip and palate.     

Preliminary results of an intelligibility measure for English-speaking children with cleft palate.

OBJECTIVE: This study describes a preliminary evaluation of the construct and concurrent validity of the Speech Intelligibility Probe for Children With Cleft Palate. DESIGN: The study used a prospective between-groups design with convenience samples. PARTICIPANTS: Participants (ages 39 to 82 months) included 5 children with cleft palate and 10 children with typical speech development and no history of craniofacial abnormalities. All children had age-appropriate language skills. INTERVENTIONS: Each child completed the Speech Intelligibility Probe for Children With Cleft Palate by imitating single words. Each child's word productions were recorded and played back to listeners who completed open-set and closed-set response tasks. Recorded utterances that represented a contiguous 100-word sample of each child's spontaneous speech also were played back to listeners for completion of an open-set word identification task. MAIN OUTCOME MEASURES: Measures reported include group means for (1) intelligibility scores for the open-set Speech Intelligibility Probe for Children With Cleft Palate and spontaneous speech sample conditions, and (2) percentage of phonetic contrasts correct and correct-distorted from the Speech Intelligibility Probe for Children With Cleft Palate closed-set response task. RESULTS: The group of children with cleft palate had significantly lower intelligibility scores, lower percentage of correct phonetic contrasts, and higher percentage of correct distorted items (construct validity). A strong positive correlation (r = .88, p < .01) was found between intelligibility scores from the Speech Intelligibility Probe for Children With Cleft Palate and the spontaneous sample (concurrent validity). CONCLUSIONS: The results provide preliminary support for the construct and concurrent validities of the Speech Intelligibility Probe for Children With Cleft Palate as a measure of children's speech intelligibility.     

Nasal deformity and microform cleft lip in parents of patients with cleft lip.

OBJECTIVE: To describe subtle nasal deformities and microform cleft lips in parents whose children have complete cleft lip deformities. DESIGN: Clinical analyses of three parents whose children had complete cleft lips. Subtle nasal deformities and microform cleft lips were identified. SETTING: An institutional general hospital: Manuel Gea González Cleft Lip and Palate Clinic in Mexico City, Mexico. PATIENTS: The study first examined the parents of all complete and incomplete cleft lip-palate patients who were seen from March 1994 to February 1997 by the authors (n = 1000). We identified three patients, each of whom had one parent who showed signs of subtle nasal deformity and microform cleft lip. INTERVENTIONS: None for the parents. Interventions in the children with cleft lips and palates were known. MAIN OUTCOME MEASURES: Qualitative photographic analyses were performed. Nostril symmetry, septal deviation, nasal floor position, and orbicularis oris malinsertions were carefully examined. RESULTS: Three of the evaluated parents had one alar cartilage caudally displaced and a deviated septum. One parent's nasal floor was depressed. Two parents also had evidence of a minimal orbicularis oris muscle fissure located in the upper lip. CONCLUSION: Genetic evaluations of children with complete and incomplete cleft lips might also include thorough evaluation of their parents. Although small in size, this study of three case histories identified nasal and lip deformities in the patients' parents.     

Effects of a child with a craniofacial anomaly on stability of the parental relationship

The purpose of this study was to determine rates of divorce in parents of children with various types of craniofacial anomalies and to analyze possible confounding factors. A 29-question survey was sent to parents of all children evaluated in the Craniofacial Centre between 1992 and 1997. Parents were questioned regarding pre- and postnatal marital stability, whether the child's facial anomaly contributed to divorce, and involvement in the child's welfare. Using deformational posterior plagiocephaly as a control group, rates of divorce vs. non-divorce were compared for craniofacial anomalies, categorized as asymmetric (hemifacial microsomia, unilateral coronal synostosis, cleft lip, cleft lip/palate) or symmetric (syndromic-craniosynostosis, orbital hypertelorism, Treacher Collins syndrome). Major anomalies (hemifacial microsomia, craniosynostosis, orbital hypertelorism, Treacher Collins syndrome) were also compared to minor anomalies (cleft lip, cleft lip/palate). Surveys were sent to both parents in 412 families; 403 surveys were returned; and the results were evaluated in 275 families (67%). Frequency analysis demonstrated an overall divorce rate of 6.8% and 4.9% separation. Anomalies associated with the highest rate of divorce were hemifacial microsomia (24.0%), syndromic craniosynostosis (12.2%), and cleft lip/palate (6.8%). 79% of non-divorced couples reported a strong prenatal relationship, whereas 59% of divorced couples reported a problematic relationship. Following birth of the affected child, 47% of non-divorced couples responded that the bonds became stronger and 41% of divorced couples thought the relationship worsened. Two-sided Fisher exact test comparing control vs. all other anomalies showed significance (p=.030) for rates of divorce. Separation of anomalies into asymmetric vs. symmetric and major vs. minor categories demonstrated no significant difference in divorce rate (p>.05). The mother was more likely to become a child's primary caregiver following estrangement.     

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.     

产前诊断唇腭裂畸形的研究进展

唇腭裂是胎儿颜面部最常见的先天性畸形,一半的病例为唇裂合并腭裂,可以孤立存在,也可合并与一些先天性畸形合并存在。由于唇腭裂畸形影响新生儿面容、吸吮、吞咽、语言及面部发育等功能,导致身心发育不良,给患儿和家庭带来不良影响。产前早期诊断胎儿唇腭裂畸形有利于孕妇得知胎儿患有唇腭裂畸形后向产科、儿科及正畸专家咨询及时作出是否继续妊娠的选择。产前超声检查是早期诊断唇腭裂最主要及最重要的筛选及诊断方法,并可进行唇腭裂分型并系统检查胎儿是否有其它器官合并畸形;胎儿磁共振对超声发现的唇腭裂特别是腭裂的诊断和严重程度评估上起辅助诊断作用。产前优生遗传检查可确定唇腭裂胎儿是否合并有染色体异常。     

Assessment of patients' level of satisfaction with cleft treatment using the Cleft Evaluation Profile.

OBJECTIVES: Determination of the psychosocial status and assessment of the level of satisfaction in Malaysian cleft palate patients and their parents. DESIGN: Cross-sectional study. PARTICIPANTS AND METHODS: Sixty cleft lip and palate patients (12 to 17 years of age) from Hospital Universiti Sains Malaysia and their parents were selected. The questionnaires used were the Child Interview Schedule, the Parents Interview Schedule, and the Cleft Evaluation Profile (CEP), administered via individual interviews. RESULTS: Patients were teased because of their clefts and felt their self-confidence was affected by the cleft condition. They were frequently teased about cleft-related features such as speech, teeth, and lip appearance. Parents also reported that their children were being teased because of their clefts and that their children's self-confidence was affected by the clefts. Both showed a significant level of satisfaction with the treatment provided by the cleft team. There was no significant difference between the responses of the patients and their parents. The features that were found to be most important for the patients and their parents, in decreasing order of priority, were teeth, nose, lips, and speech. CONCLUSIONS: Cleft lip and/or palate patients were teased because of their clefts, and it affected their self-confidence. The Cleft Evaluation Profile is a reliable and useful tool to assess patients' level of satisfaction with treatment received for cleft lip and/or palate and can identify the types of cleft-related features that are most important for the patients.     

Community orthodontists and craniofacial care: results of a Washington state survey.

BACKGROUND: Little is known about community orthodontists' previous training in, experience with, or receptivity to caring for children with craniofacial disorders. OBJECTIVES: (1) To characterize the current level of participation by Washington state orthodontists in craniofacial care; and (2) to identify factors that promote or impede community orthodontists' involvement in caring for children with craniofacial conditions. DESIGN: Mail survey. METHODS: A 26-item questionnaire was designed and mailed to all active orthodontists in Washington state (N = 230). Question topics included practice characteristics, training and experience with craniofacial conditions, concerns related to public and private insurance, and communication with craniofacial teams. RESULTS: Of eligible respondents, 68% completed the survey. Most orthodontists' patient panels were made up of patients who either have private insurance or pay cash for services. On average, 2% of respondents' patients were Medicaid beneficiaries. Only 20% of respondents had seen more than three patients with cleft lip and/or palate in the past 3 years. Although a minority of orthodontists receive referrals from (27%) or are affiliated with (11%) craniofacial teams, most orthodontists perceived craniofacial care positively and were interested to learn more about craniofacial care and to accept additional patients with these conditions. CONCLUSIONS: Results of this survey can inform potential strategies to increase access to orthodontic care for children with craniofacial disorders. These would include developing an organized training, referral, and communication system between community orthodontists and state craniofacial teams and considering a case-management approach to facilitate this process.     

The craniofacial morphology of the parents of children with orofacial clefting: a systematic review of cephalometric studies

Abstract

Objective To systematically review the cephalometric studies investigating the craniofacial morphology of the parents of children with orofacial clefting (OFC).

Search strategy The search strategy was based on the keywords ‘parent‘, cephalometry', and ‘cleft‘, identifying 17 studies, of which 15 ‘case/control’ studies met the inclusion criteria

Statistically significant clinically relevant cephalometric variables from univariate statistical tests and multivariate results were collated and presented unweighted.

Results/Conclusions The parental craniofacial complex in OFC is distinctive in comparison to the non-cleft population. However, there is insufficient consistency in study designs and results to accurately characterize the parents of children with OFC. Although the craniofacial morphology of the parents of children with CL(P) differs to the parents of children with CP, there is insufficient information to accurately localize these differences.     

Beyond easy answers: the plastic surgeon and prenatal diagnosis.

OBJECTIVE: To explore how plastic surgeons handle the issue of prenatal diagnosis of cleft lip and palate. DESIGN: Mail survey. PARTICIPANTS: The plastic surgeon listed as team contact in the team directory of the American Cleft Palate-Craniofacial Association was identified. In cases in which a plastic surgeon was not listed as a team contact, the first plastic surgeon listed as a team member was identified. A total of 211 surgeons were contacted. INTERVENTION: Each surgeon was mailed a survey asking for his/her experience in prenatal diagnosis for cleft conditions. Ethical issues concerning the surgeon's personal beliefs about abortion and the potential influence of the surgeon's presentation of information were explored. RESULTS: A 50% response rate was obtained. Eighty-five percent of respondents were experienced in prenatal counseling; 92% offer consults on a routine basis. Ninety-six percent indicated they did so because they felt it was helpful to families. Treatment (98%), appearance of clefts (96%), cleft etiology (94%), and associated problems (40%) were discussed most commonly. Termination was rarely discussed (5%). Of those who had not met with a family, 82% said they had never been asked to do so. Most respondents felt families sought consultation for information and rarely sought consultation to decide on abortion. The majority felt their personal beliefs about abortion did not influence their willingness to meet with parents. The majority of surgeons in both groups felt it was ethical to give an opinion on pregnancy termination if asked (55% of experienced surgeons, 44% of inexperienced surgeons); however, a significantly larger number of the surgeons who had not met with a family were unsure of their opinion. CONCLUSIONS: Prenatal counseling of parents of a fetus with cleft lip/palate is common among plastic surgeons. There is uniformity in the information discussed. There is no agreement among surgeons about the ethical issues of pregnancy termination and the surgeon's role in that discussion.     

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。     

A study of cephalometric features in cleft lip-cleft palate families. I: Phenotypic heterogeneity and genetic predisposition in parents of sporadic cases

Several previous studies have indicated that unaffected parents of children with nonsyndromic cleft lip and palate show unusual craniofacial features. This study reexamines this question by applying multivariate cluster analysis to lateral cephalometric head plates from 82 individuals who are parents of sporadic cases of cleft lip with or without cleft palate (CL/P). Considerable phenotypic heterogeneity was present within the sample. Three major groupings were defined. Two of these groups showed cephalometric similarities to individuals with overt clefts, while the third showed a generalized concordance to published norms. In almost every case only one member of each parental pair showed the cleft related cephalometric phenotype, suggesting the possibility of a substantial genetic component in many cases of sporadic CL/P. However, there were several cases in which neither parent showed the phenotypic traits. Such cases may have a different etiology or a greater environmental component.     

Parental craniofacial cephalometric features of Chinese patients with nonsyndromic isolated cleft palate

To evaluate the parental craniofacial morphology in Chinese patients with sporadic nonsyndromic cleft palate, 175 parental pairs of children with nonsyndromic cleft palate (NSCP) and 206 controls from Sichuan University were involved in this study. Conventional cephalometric analysis was used to measure angles, linear distances and their ratios. Two-sample Student's t-tests and multivariate discriminant analysis were applied to the data. The data indicated that fathers of children with NSCP tended to have longer anterior cranial base length, palatal length, mandibular body length, mandibular ramus length, and longer mandibular height, lower face height, as well as larger nasal width, palatal width, mandibular body width, and condyle distance (p < 0.05). Mothers of children with NSCP tended to have longer mandibular body length, mandibular ramus length, and longer mandibular height, lower face height, as well as larger nasal width, palatal width, mandibular body width, and condyle distance (p < 0.05). The results indicated that the healthy parents of the patients with NSCP show distinct characteristics in craniofacial morphology. In general, the characteristics seem to be more distinct in the fathers of cleft patients than in the mothers.     

Family environment of individuals with oral clefts in Argentina.

OBJECTIVE: The purpose of this investigation was to study the social environment of families of children with different types of nonsyndromic oral clefts (OC) and to compare these groups with a control population of families of children without clefts. DESIGN: The study compared three nonsyndromic oral cleft groups and the control group using the Moos Family Environment Scale, which examines cohesion, expressiveness, conflict, independence, achievement-orientation, intellectual-cultural orientation, active-recreational orientation, moral-religious emphasis, organization, and control. SETTING: All parents of children with nonsyndromic oral clefts from a large craniofacial clinic in Buenos Aires, Argentina, were identified and were enrolled in this study between June 2000 and August 2001. Control families were ascertained from the pediatrics service of a hospital located in the vicinity of the craniofacial clinic. PARTICIPANTS: One hundred and sixty-five parents were selected, based on having a child with nonsyndromic unilateral cleft lip with or without cleft palate (UCL/P), bilateral cleft lip with or without cleft palate (BCL/P), or isolated cleft palate (ICP). One hundred and eighty control parents with no family history of congenital anomalies were selected, as well. RESULTS: There was no major difference in the social environment of families of children with different types of nonsyndromic oral clefts. When compared with families in the control group, families of children with nonsyndromic oral clefts scored better in all three subdimensions of family relationship, revealed a high level of independence, and showed better structure and organization than control families did; however, families of children with nonsyndromic oral clefts reported participating in fewer recreational activities. CONCLUSIONS: Overall, families of children with nonsyndromic oral clefts displayed a good social environment. Efforts should be focused to involve them in recreational activities.