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PubMed2005

Human prenatal nasal bone lengths: normal standards and length values in fetuses with cleft lip and cleft palate.

OBJECTIVE: The aim of this study was to present prenatal autopsy standards for nasal bone length in normal fetuses, as well as to compare nasal bone lengths in human fetuses with cleft lip and palate with those standards. MATERIAL AND METHODS: The material consisted of human fetuses investigated in connection with diagnosed and legally approved abortions and spontaneous abortions; 40 were normal and 26 had cleft lip and palate (7 isolated cleft lip, 12 isolated cleft palate, and 7 combined cleft lip and palate). Menstrual ages (MA) ranged from 12 to 25 weeks, crown-rump length (CRL) from 55 to 210 mm and foot length (FL) from 7 to 44 mm. All fetuses were radiographed in lateral projection, and, using these images, the nasal bone lengths were measured with a digital caliper. Regression analyses were performed. RESULTS: Standards for normal nasal bone length at different ages are expressed as menstrual age, crown-rump length, or foot length. Comparisons with normal standards showed that the isolated cleft lip group had a significantly smaller nasal bone length than the normal fetuses had. Fetuses with isolated cleft palate and fetuses with combined cleft lip and palate showed no significant deviation from normal standards. CONCLUSION: Normal nasal bone standards can contribute to information on deviations in nasal bone length for aborted fetuses with known and unknown genotypes. The differences in nasal bone lengths in fetuses with different lip and palate clefting can contribute to understanding the pathogenesis of cleft lip and palate.

Aborted Fetus↗
PubMed1978

Genetic counselling and genetics of cleft lip and cleft palate.

Modern neonatal care and advanced plastic surgical correction have led to the survival of most newborns with oral clefts. These children are likely to reproduce. A slight increase in the incidence of oral clefts may be expected in the future. The genetics of cleft lip and cleft palate is reviewed. The inheritance is usually multifactorial. With normal parents the risk of having a first affected child with cleft lip is about one per thousand, the risk of having a second affected child 4 per cent and the risk of having a third affected child 10 per cent. If a parent has already a cleft lip, the risk of having a first affected child now is 4 per cent, while the risk of having a second affected child is 10 per cent. The methodology of genetic counseling is given.

Abnormalities, Drug-Induced↗
PubMed1975

Growth and changes in maxillary arch form in complete unilateral cleft lip and cleft palate children.

This study aims at the comparison of growth changes of the maxillary arch of 62 normals and 87 complete unilateral cleft lip and cleft palate subjects. This is achieved by measuring their maxillo-facial models, which will aid in clarifying such questions as: when and where the maxillary growth inhibition will occur, and the several aspects of growth-change. Patients with complete unilateral cleft lip and cleft palate were classified into four stages: 1) six month old infants before lip and palatal closure; 2) two year old children, lip repair at six months of age; 3) three year old children, lip repair at six months and palatal closure at two years; 4) four year old children, with repairs as in stage 3. Normal subjects were also classified into four stages, to match the cleft group by age and body weight. Impressions were taken for each patient's upper jaw and upper face simultaneously under general anesthesia, and the maxillo-facial model was made. Results obtained were as follows: (1) In the normal group, the forward and downward growth of the anterior alveolar region increased considerably between stages 1 and 2 and stages 3 and 4. The growth of the anterior alveolar arch was slight during all four stages. The depth and width of the retromolar point increased gradually through all four stages, and measured the growth of the posterior alveolar region. (2) In the cleft group: at stage 1 the anterior end of the larger segment was protruded and both the larger and smaller segments, especially in the anterior region, were laterally dislocated. The height of the anterior alveolar region was found to be noticeably less than that of the normal group. At stage 2 the growth inhibition in depth and height was noted in all the alveolar points of the cleft group, especially in the anterior alveolar region, in comparison to the normal group. At stage 3 the depths in all alveolar points decreased, but no significant difference was found when compared to stage 2. However, the increase in heights was marked. At stage 4 the depths and heights of alveolar points in the cleft group were found to be smaller than those of the normal group.

Child, Preschool↗
PubMed2006

[Alpha-tocopherol levels in mothers of children with cleft lip or with cleft lip and palate].

OBJECTIVES: Environmental factors may play equally critical role as genes in facial embryogenesis. It was suggested that cleft palate occurring in association with cleft lip may result from environmental factors that affect development of both the lip and the palate. Cleft lip forms from the third through seventh week of embryonic development. During these period fetal nutrition is histotrophic, pressure of oxygen is low, genes for antioxidant enzymes are supressed and alpha-tocopherol play an essential role in the fetal tissues antioxidant capacity. Cleft palate forms from the fifth through twelfth weeks of gestation, when hemonutrition and antioxidant enzymes are developing, and hypothetical deficiency of tocopherol is not so crucial. AIM: The goal of the study was to investigate concentrations of alpha-tocopherol in women who gave birth to a child with cleft lip and mothers of children with cleft lip and palate. MATERIAL AND METHODS: Blood samples from 31 healthy mothers of children with isolated cleft lip (CL) and 29 healthy mothers of children with isolated cleft lip and palate (CLP) were collected. Fasting plasma alpha-tocopherol concentrations were measured by the high performance liquid chromatography. Kolmologorov-Smirnov test with Lilefors correction and k-means cluster analysis were applied to determine differences between the groups of women. RESULTS: We found that alpha-tocopherol concentrations were lower in CL compared to CLP, but the difference did not achieve level of statistical significantly (medians: 15.8 micromol/L vs. 20.0 micromol/L; p = 0.066). There were identified three clusters of alpha-tocopherol concentrations: 14.0 micromol/L (19CL+8CLP), 19.6 micromol/L (8CL+13CLP), and 21.1 micromol/L (4CL+8CLP). The distribution of results to the clusters was dependent on type of the orofacial cleft (chi squared = 6.95 with 2 degrees of freedom, p = 0,031). CONCLUSIONS: Analysis for environmental risk factors should be performed separately for cleft lip and cleft lip and palate because these two conditions appear to be etiologically distinct. The role of a higher periconceptional intake of alpha-tocopherol in reduction of risk of giving birth to a child with cleft lip requires further research.

Adult↗
PubMed1998

[An epidemiological survey of cleft lip and cleft palate in Fujian province].

OBJECTIVE: To find out the incidence of cleft lip and cleft palate in Fujian provice and to analysis the factors of its deformity. METHODS: From Oct. 1986 to Dec. 1992, the birth defect monitoring was carried out in 22 hospitals in Fujian province. RESULTS: It was found that there were 204 cases of cleft lip (CL) and cleft palate (CP) in 139,882 perinatals. The incidence rate of CL and/or CP was 1.46@1000. Annual incidence rate of CL and/or CP had not found any significant change. CONCLUSION: Among the 204 cases 28 had other deformities. The incidence rate of CL and/or CP related to maternal age, birth order, smoking factors were studied.

Adolescent↗
PubMed2000

Comparative study between children with and without cleft lip and cleft palate, part 1: cephalometric analysis.

OBJECTIVE: This study was conducted to compare craniofacial relationships, position, and curvature of the cervical spine between children with cleft lip and cleft palate who had been operated on and children without clefts. METHOD: This study was performed in 28 children with mixed dentition. They were divided into two groups. The study group included 14 children with unilateral operated cleft lip and cleft palate, ranging in age from 6 to 12 years, who clinically presented with a short upper lip, abnormal lip seal, and inhibition of sagittal development of the midface that was radiographically assessed. The control group included 14 children without clefts, ranging in age from 8 to 11 years. All of them had normal lip seal, nasal breathing, and a clinically normal body posture. DESIGN: A lateral craniocervical radiograph in a self-balanced natural head position in an erect posture, and without using a head holder, was taken for each child of both groups, with the mandible in maximum intercuspation and lips in habitual posture. The true vertical was marked on all the films. Specific angular and linear dimensions were used to assess the craniocervical relationships, as were the position of the cervical spine, its curvature, or both. RESULTS AND CONCLUSIONS: The study group presented a significant increase in the extension of the head on the neck, forward position of the cervical spine, and a decrease in the curvature of the cervical spine in comparison with the children without clefts. These results are more relevant considering that the study group also presented higher significant values of lower facial height than children without clefts.

Cephalometry↗
PubMed1993

Mandibular growth in patients with cleft lip and/or cleft palate--the influence of cleft type.

A total of 229 adult white patients with cleft lip or palate were evaluated, through cephalometric radiographs, to determine cleft-type influence on mandibular structure and spatial position. The sample includes adult patients with unilateral complete cleft lip and alveolus (n = 50), complete cleft lip and palate (n = 118), and isolated cleft palate (n = 61). A group of 65 subjects with normal occlusion and without cleft lip or palate was used as a control group. All groups were matched according to sex. When compared, the cleft groups and the normal group showed that the mandibular structure was significantly different. The mandible in the cleft groups displayed shorter mandibular ramus and body length, without difference between the cleft types. Mandibular position relative to cranial base was similar in the cleft groups with involvement of the palate, and differed significantly from that observed in the group with cleft lip and alveolus and in the subjects without cleft. Palatal clefts induce a significant downward and backward rotation of the mandible associated with a more obtuse gonial angle. Thus a small mandible is a structural characteristic of all cleft types studied. However, mandibular shape and spatial position are strongly influenced in clefts that involve the palate.

Adolescent↗
PubMed1993

[The prevalence and risk factors of cleft lip and cleft palate in 2 hospitals in the city of Guadalajara, Jalisco, Mexico].

OBJECTIVE: The purpose of this article is to determine the prevalence of the cleft lip and palate and its association with some risking factors. MATERIAL AND METHODS: A retrospective, observational and analytic study, of 44 newborn infants with cleft lip and palate and their controls, in 33,461 consecutive births, with 20 weeks or more, and birth weight more than 500 g, since November of 1988 to June of 1991. The information were obtained from the database of the Congenital External Malformation Register, done by the University of Guadalajara, in four hospitals. The prevalence of the cleft lip and palate was calculated. Of the variables studied their association was searched with congenital malformation in 2 x 2 tables, calculating, its chi square, confidence interval, and old ratio. RESULTS: The prevalence of the cleft lip and palate was of 13.2 x 10,000 births. By categories: cleft lip 2.7 x 10,000, cleft lip and palate 7.8 x 10,000 and cleft palate 2.7 x 10,000. The only statistics differences with control group were the association with antecedent of other malformation in the family and methrorrague during pregnancy. CONCLUSIONS: The found prevalence is similar to the one informed in the literature, with some differences by categories. The association with antecedent of another malformation in the family, support the role of the hereditary role in the multifactorial etiology.

Chi-Square Distribution↗
PubMed1989

[Van der Woude syndrome (lip pits, cleft lip and cleft palate syndrome)].

Three patients with lip pits among 701 cases (0.43%) with clefts were treated in Beijing Stomatological Hospital in last five years. Genetic analysis of cases in 5 families with Van der Woude syndrome (Lip pits, cleft lip and cleft palate) in China was studied. We report on three unrelated families from the Beijing area, other three families from the Hebei, Heilongjang, Gansu province, in which the autosomal dominant gene for Van der Woude syndrome is segregating. All three or one of the clinical features may occur in an affected individual who carries the gene. Two probands had ankyloglossia. Six members were missing upper lateral incisors. One proband was missing second premolars. Six members were cross-bite. Two probands had congenital fistula of the mouth angle.

Child↗
PubMed1993

Analyses of craniofacial and dental morphology in monozygotic twins discordant for cleft lip and unilateral cleft lip and palate.

The Michigan Cleft Twin Sample data was analyzed in order to investigate dental arch and craniofacial morphology. The present study consisted of 12 monozygotic twins discordant for either cleft lip or unilateral cleft lip and palate. Individuals having CL (repaired) alone did not differ from their unaffected counterparts in dental arch and skeletal morphology. Repaired UCLP subjects were characterized by having a shorter and more posteriorly positioned maxilla. Although reduction of dental arch width as a result of scar contraction following surgery was evident, the effects of scar tissue pull did not affect maxillary basal width.

Cephalometry↗
PubMed1995

Augmentation of the free border of the lip in cleft lip patients using temporoparietal fascia.

Deficiency of the free border of the lip is frequently encountered in secondary cleft lip deformities. The free border of the lip consists of the orbicularis oris marginalis muscle and its overlying subcutaneous tissue, vermilion, and mucosa. The pathology may involve any or all of these components. When there is an absolute shortage of subcutaneous tissue or orbicularis oris marginalis muscle, extra tissue from another source must be used for augmentation. A temporoparietal fascial graft is a simple and reliable method to increase the fullness of the free border of the lip. The tissue is soft in consistency and displays minimal resorption. Most important, more complicated procedures such as an Abbé flap or tongue flap may be avoided. From July of 1988 until June of 1992, the temporoparietal fascial graft was used successfully to correct deficiencies of the free border of the lip in 20 patients with cleft lip deformities. There were two graft exposures that healed with conservative treatment. The procedure is a useful method of augmenting both lateral lip and central lip deficiencies in secondary and tertiary cleft lip deformities.

Adolescent↗
PubMed2003

Incidence and outcome of middle ear disease in cleft lip and/or cleft palate.

OBJECTIVE: Otitis media with effusion is known to be very common among children with cleft palate, however, less is known regarding the natural history and outcome in this group. The purpose of the present study was to examine the incidence, natural history, treatment, and outcome of middle ear disease in children with clefts. METHODS: A questionnaire was sent to the parents of all children registered on the cleft lip and palate database at our institution. The medical records of all respondents were also reviewed. Statistical analysis of the results was performed using Fisher's exact test in contingency tables and binary logistic regression analyses, where appropriate. RESULTS: 397 fully completed questionnaires were returned. Ear disease was much more common in children with cleft palate, or cleft lip and palate, than in children with cleft lip. Among children with cleft palate, ear problems (infections and/or hearing loss) were most prevalent in the 4-6-year-old age group. However, ear problems persisted at a substantial level for many years after this; only after the age of 12 years did problems appear to settle. The incidence of below normal current hearing and of surgery for chronic otitis media was significantly related to history of ear infections (P=0.000 and 0.000, respectively), and to increased number of ventilation tube insertions (P=0.000 and 0.000, respectively). CONCLUSIONS: Middle ear disease is common in children with cleft palate, and, unlike the case for children without clefts, has a prolonged recovery, and a substantial incidence of late sequelae. The higher incidence of below normal hearing and surgery for chronic otitis media in children undergoing a greater number of ventilation tube insertions, although most likely reflecting an increased underlying severity of otitis media in these children, also underlines the lack of long-term benefits of ventilation tubes in this group.

Adolescent↗
PubMed1980

Summary of a conference on cleft lip and cleft palate.

Although different opinions exist about surgical repair of cleft lip and cleft palate, cooperation between a number of specialities is essential to a successful result and final rehabilitation of the patient.

Alveoloplasty↗
PubMed1983

Ethnic differences in the incidence of cleft lip and/or cleft palate in Auckland, 1960-1976.

The incidence of facial clefts among live births in the Auckland urban area has been studied for the years 1960-1976. After correction for ascertainment, the live-born incidence of non-syndromal cleft palate among Maoris was 1.867/1000, and among Europeans it was 0.643/1000. The incidences for non-syndromal cleft lip, with or without cleft palate, were 0.397/1000 for Maoris, and 1.195/1000 for Europeans. The sex ratio for cleft palate showed the usual female preponderance, although there was evidence of heterogeneity between the races. Cleft lip occurred more commonly among males. Birth rank, maternal age, and paternal age had no apparent effect on the incidences, and there were no significant secular or seasonal changes in incidence.

Cleft Lip↗
PubMed1982

Male-to-male transmission of Robinow's syndrome. Its occurrence in association with cleft lip and cleft palate.

The inheritance of Robinow's syndrome has been thought to be autosomal dominant. However, since no cases of male-to-male transmission have been reported, it has been impossible to rule out X-linked dominant inheritance. We studied a case of male-to-male transmission in a father and son with Robinow's syndrome and cleft lip-cleft palate, confirming autosomal dominant inheritance in at least some cases.

Abnormalities, Multiple↗
PubMed1987

Walker-Warburg syndrome with cleft lip and cleft palate in two sibs.

Two sibs are reported with Walker-Warburg syndrome including hydrocephalus, agyria, anterior chamber dysgenesis, and encephalocele. In addition, both had cleft lip and cleft palate and intrauterine growth retardation, findings not previously noted in this condition.

Abnormalities, Multiple↗
PubMed2005

Agenesis of tibia with bifid femur, congenital heart disease, and cleft lip with cleft palate or tracheoesophageal fistula: possible variants of Gollop-Wolfgang complex.

Two patients with bifurcation of one femur, ipsilateral absence of tibia and one partially formed tibia, or bilateral absent tibiae, club feet with normal or nearly normal digits, congenital heart disease and, in one case, tracheoesophageal fistula; in the second, cleft lip and cleft palate and enlarged head with increased fluid and lissencephaly, are described. These appear to be unique combinations of defects but overlap with the Gollop-Wolfgang complex is present, particularly with the case of possible Gollop-Wolfgang described by Raas-Rothschild et al.

Abnormalities, Multiple↗