An investigation of the dentition of parents of children with cleft lip and palate.

OBJECTIVE: The aim of this investigation was to identify whether any dental features in parents of children with cleft lip and palate can be useful as predictors of clefting. METHODS: The dentition of 28 pairs of parents of children with complete unilateral (17) and bilateral (11) cleft lip and palate and 21 pairs of parents of noncleft children were evaluated. Clinical and radiographic examinations were carried out to identify abnormalities of tooth number and morphology. Study cast assessment was undertaken to evaluate incisor relationship, overjet, overbite, intercanine widths, and mesiodistal widths of individual teeth, and these data were subjected to statistical analysis. RESULTS: The prevalence of abnormalities of tooth number in parents of cleft children was similar to those reported elsewhere for general populations. In parents of children with unilateral clefts, there was no tooth-size asymmetry between teeth on the side corresponding to the child's cleft and noncleft sides, respectively. No differences were found in tooth widths and intercanine widths between parents of children with unilateral clefts and parents of children with bilateral clefts. The incisor relationship, overjet, overbite, and intercanine widths were found not to differ statistically between the parents of cleft children and parents of noncleft children. CONCLUSIONS: No predictors of clefting could be identified in the dentition of parents of children with cleft lip and palate.     

An epidemiologic study of oral clefts in Iran: analysis of 1,669 cases.

OBJECTIVE: The objective of this study was to assess the epidemiology and some genetic aspects of oral clefting in Iran. DESIGN: The study was a 15-year cross-sectional (prevalence) study from August 1976 to September 1991. SETTING: The setting for the study was two plastic surgery departments, both mostly referral centers, and a maternity hospital in Iran. PARTICIPANTS: The participants were 1,669 consecutive surgical cases with oral clefts (79% between 1 day and 18 months of age), registered in two centers. In a parallel study, 19,369 livebirths born in a maternity hospital within the same period were surveyed to ascertain prevalence of clefting at birth. Normal children of this population were used as the control group. Samples were analyzed by prevalence of clefting at birth, type of oral cleft, side of clefting, sex ratio, associated malformations, clefts in relatives, and parental consanguinity. RESULTS: The prevalence of clefts was 1.03 per 1,000 births. Cleft lip (without cleft palate) had a higher (34.9%) and cleft palate alone had a significantly lower prevalence (17.4%) than expected. Cleft lip (without cleft palate) was more common in females (53.6%) than in males (46.4%). The rate of associated malformations in cleft patients (7.73%) was higher than in controls (0.093%). Association between clefting and consanguinity was significant (45.8% in cases versus 43.1% in controls). Occurrence of noncleft congenital malformations among first-degree relatives of our cases (2.77%) was nearly two times that of controls (1.55%). CONCLUSION: Our study reveals that the population incidence of oral clefts in Iran is much closer to European than Arab-African or South East Asian countries. However, significant variations in other epidemiologic and some genetic features were observed.     

The incidence and relationship of cervical spine anomalies in patients with cleft lip and/or palate

The relationship, incidence, and distribution of cervical spine anomalies were assessed in 468 patients with cleft lip and/or palate. The patients were placed into four groups: lip and/or alveolar; complete unilateral or bilateral; isolated palatal; and soft palate or submucous clefts. Cervical anomalies were observed in 22% of the cleft patients and in 7% of the noncleft group. Patients with soft palate and submucous clefts had the highest incidence of vertebral anomalies (45%), whereas patients with cleft lip and/or alveolus had an incidence similar to the noncleft group. Patients with complete unilateral and bilateral clefts also had a higher incidence (15.6% to 19.0%) of anomalies than the noncleft group. Cervical anomalies occurred primarily in the occipital-C1-C2 region. The possible implications of these findings are discussed.     

Determination of facial symmetry in unilateral cleft lip and palate patients from three-dimensional data: technical report and assessment of measurement errors.

OBJECTIVE: To assess measurement errors of a novel technique for the three-dimensional determination of the degree of facial symmetry in patients suffering from unilateral cleft lip and palate malformations. DESIGN: Technical report, reliability study. SETTING: Cleft Lip and Palate Center of the University of Erlangen-Nuremberg, Erlangen, Germany. PATIENTS: The three-dimensional facial surface data of five 10-year-old unilateral cleft lip and palate patients were subjected to the analysis. Distances, angles, surface areas, and volumes were assessed twice. MAIN OUTCOME MEASURES: Calculations were made for method error, intraclass correlation coefficient, and repeatability of the measurements of distances, angles, surface areas, and volumes. RESULTS: The method errors were less than 1 mm for distances and less than 1.5 degrees for angles. The intraclass correlation coefficients showed values greater than .90 for all parameters. The repeatability values were comparable for cleft and noncleft sides. CONCLUSION: The small method errors, high intraclass correlation coefficients, and comparable repeatability values for cleft and noncleft sides reveal that the new technique is appropriate for clinical use.     

Dental age in children with a complete unilateral cleft lip and palate.

OBJECTIVE: To assess dental age in children with a complete unilateral cleft lip and palate and to compare this with a noncleft control group. DESIGN: Two-group, mixed-longitudinal cohort study. SETTING: Cleft group from an academic center for cleft lip and palate treatment. Noncleft control group from the same population. PATIENTS: Participants included 70 Caucasian children with a full complement of teeth and a complete unilateral cleft lip and palate (45 boys and 25 girls) from the Cleft Palate Craniofacial Center at the Radboud University Nijmegen Medical Center, Nijmegen, the Netherlands. The control group (90 boys and 91 girls) was taken from the Nijmegen Growth Study. MAIN OUTCOME MEASURE: Dental age was assessed on orthopantomograms. In the unilateral cleft lip and palate group, linear interpolation in individual age curves was applied to obtain the dental age at 5, 9.5, and 14 years of age. For these ages, a comparison was made with the noncleft control group. RESULTS: Boys and girls with a unilateral cleft lip and palate showed a significant delay in dental age, as compared with their noncleft peers at all three ages. This delay was more pronounced in boys than in girls. The gender effect was significant at chronological ages 5 and 14 years. CONCLUSIONS: Children with a complete unilateral cleft lip and palate have a delay in dental age, compared with noncleft children.     

Data-objective diagnosis of infant cleft lip, alveolus, and palate. Morphologic data guiding understanding and treatment concepts

Maxillary plaster models of untreated infants with various cleft lip and palate forms have been studied under the Reflex Microscope. From the three- dimensional data processed by a PC, the specific dimensions of various cleft forms can be determined. Unexpected findings are that skeletal maxillary changes are present in infant velar clefts. The mid-sagittal maxillary length in particular becomes shorter with more severe clefting of the bony palate. From the morphologic point of view, Robin's sequence does not represent a specific entity, but usually a severe cleft of the hard palate. Unilateral clefts of lip, alveolus, and palate with a partial cleft lip reveal worse malformations than complete unilateral clefts. The morphologic changes recorded can be explained by a varying degree of lingual malposition. This reveals that displacements and malformations of infant cleft lip and palate are in fact secondary in nature. In correcting these secondary surface manifestations as soon as indicated by physiologic criteria one could hope for early normalization of sensation, perception, and function. Three-dimensional high-precision measurements are crucial for data-objective diagnoses, timing of therapy, choice of treatment, and in later evaluation of the results.     

Prevalence of enamel hypoplasia in deciduous canines of patients with complete cleft lip and palate.

OBJECTIVE: To evaluate the prevalence and type of enamel hypoplasia in deciduous canines of patients with complete unilateral and bilateral cleft lip and palate, as well as its distribution according to gender, dental arch, and side of cleft. DESIGN: Cross sectional. SETTING: Hospital for Rehabilitation of Craniofacial Anomalies, Bauru, S?o Paulo, Brazil. MATERIAL AND METHODS: Three-hundred twelve Caucasian children (193 boys, 119 girls), aged 3 to 10 years, presenting complete unilateral and bilateral cleft lip and palate were included in this study. A single examiner carried out clinical examination under natural light with a dental probe and dental mirror and, after drying of the tooth, by means of a simplified hypoplasia index. RESULTS: A prevalence of 43.8% was observed for unilateral and 39% for bilateral clefts. Both groups revealed similar distributions regarding the maxillary and mandibular dental arches and gender. Similar distribution was also observed on the cleft and noncleft sides for the unilateral cleft group. The most frequent type and severity was obvious hypoplasia, followed by minimal hypoplasia, both roughly round. CONCLUSION: The results suggest that the cleft does not influence the occurrence of hypoplasia in deciduous canines.     

Cranial base morphology in cleft lip and palate: a cephalometric study from 7 to 18 years of age.

Four cleft lip/palate groups were cephalometrically compared with a noncleft control group using three cranial base dimensions: anterior (ACB) and posterior (PCB) cranial base, and cranial base angle (CBa). The groups were compared by age from 7 to 18 years. The results showed that the cleft lip and/or alveolus group was not different from the control group in the three dimensions. Unilateral and bilateral cleft lip and palate and isolated cleft palate cases were generally shorter in ACB and PCB for most of the growth period, particularly after age 8 to 9 years. The cranial base angle remained similar for all the cleft groups when compared with the noncleft group. These findings support the view that intrinsic tissue defects or errors during embryologic development may result in basicranial abnormalities and orofacial clefting.     

Visual and statistical modeling of facial movement in patients with cleft lip and palate.

OBJECTIVE: To analyze and display facial movement data from noncleft subjects and from patients with cleft lip and palate by using a new dynamic approach. The hypothesis was that there are differences in facial movement between the patients with cleft lip and palate and the noncleft subjects. SETTING: Subjects were recruited from the University of North Carolina School of Dentistry Orthodontic and Craniofacial Clinics. PATIENTS, PARTICIPANTS: Sixteen patients with cleft lip and palate and eight noncleft "control" subjects. INTERVENTIONS: Video recordings and measurements in three dimensions of facial movement. MAIN OUTCOME MEASURES: Principal component (PC) scores for each of six animations or movements and dynamic modeling of mean animations. STATISTICS: Multivariate statistics were used to test for significant differences in the PC mean scores between the patient groups and the noncleft groups. RESULTS: No statistically significant differences were found in PC mean scores between the patient groups and the noncleft groups; however, the variability of the effect of clefting on the soft tissues during animation was noted when the noncleft data were used to establish a "normal" scale of movement. Compensatory movements were seen in some of the patients with cleft lip and palate, and the compensation was not unidirectional. CONCLUSION: Measures of mean movement differences as summarized by PC scores between patients with cleft lip and palate and noncleft subjects may be misleading because of extreme variations about the mean in the patient group that may neutralize group differences. It may be more appropriate to compare patients to a noncleft normal scale of movement.     

Genetic association studies of cleft lip and/or palate with hypodontia outside the cleft region.

OBJECTIVE: The purpose of this study was to determine whether the candidate genes previously studied in subjects with cleft lip, cleft palate, or both are associated with hypodontia outside the region of the cleft. SUBJECTS: One hundred twenty subjects from the Iowa Craniofacial Anomalies Research Center were selected based on the availability of both dental records and genotype information. METHOD: The type of orofacial clefting and type and location of dental anomalies (missing teeth, supernumerary teeth, or peg laterals) were assessed by dental chart review and radiographic examination. Genotype analysis of candidate genes was performed using polymerase chain reaction/single-strand conformation polymorphism analysis. RESULTS: The prevalence of hypodontia in this sample was 47.5%, with 30.0% of subjects having missing teeth outside the cleft. There was a positive association between subjects with cleft lip or cleft lip and palate who had hypodontia outside the cleft region (compared with noncleft controls) and both muscle segment homeo box homolog 1 (MSX1) (p =.029) and transforming growth factor beta 3 (TGFB3) (p =.024). It was not possible in this analysis to determine whether this association was specifically associated with orofacial clefting combined with hypodontia or whether it was due primarily to the clefting phenotype. CONCLUSIONS: In this sample, there was a significantly greater incidence of hypodontia outside the cleft region in subjects with cleft lip and palate, compared with cleft lip only or cleft palate only. Cleft lip and/or palate with hypodontia outside the cleft region was positively associated with both TGFB3 and MSX1, compared with noncleft controls.     

Associations between severity of clefting and maxillary growth in patients with unilateral cleft lip and palate treated with infant orthopedics.

OBJECTIVE: The purpose of this study was to examine possible associations between severity of clefting in infants and maxillary growth in children with complete unilateral cleft lip and palate. DESIGN: This was a retrospective study of measurements made on infant maxillary study casts and maxillary cephalometric variables obtained at 5 to 6 years of follow-up. SETTING: The study was performed at the Institute of Reconstructive Plastic Surgery of New York University Medical Center, New York, New York. PATIENTS: Twenty-four consecutive nonsyndromic unilateral complete cleft lip and palate patients treated during the years 1987 to 1994. INTERVENTIONS: All the patients received uniform treatment (i.e., presurgical orthopedics followed by gingivoperiosteoplasty to close the alveolar cleft combined with repair of the lip and nose in a single stage at the age of 3 to 4 months). Closure of the palate was performed at the age of 12 to 14 months. RESULTS: Infant maxillary study cast measurements correlated in a statistically significant manner with maxillary cephalometric measurements at age 5 to 6 years. CONCLUSIONS: The results demonstrate the large variation in the severity of unilateral cleft lip and palate deformity at birth. Patients with large clefts and small arch circumference, arch length, or both demonstrated less favorable maxillary growth than those with small clefts and large arch circumference or arch length at birth.     

Early assessment of dental arch development in repaired unilateral cleft lip and unilateral cleft lip and palate versus controls.

OBJECTIVE: To evaluate dental arch relationships and dimensions, relative to an age matched noncleft sample, in Caucasian 3-year-old children with repaired unilateral cleft lip (UCL) or unilateral cleft lip and palate (UCLP). DESIGN: Prospective, cross-sectional, case-control study performed in Scotland, U.K. PARTICIPANTS: Eleven children with repaired unilateral cleft lip, 16 children with repaired unilateral cleft lip and palate, and 78 children as controls. MAIN OUTCOME MEASURES: Dental arch relationships and linear arch dimensions. RESULTS: Prevalence of Class III incisor relationship was 31.3% in children with unilateral cleft lip and palate compared with 9.1% in children with unilateral cleft lip. A buccal crossbite was present in 36% of children with unilateral cleft lip, compared with 75.6% of children with unilateral cleft lip and palate.Mean linear maxillary arch dimensions did not differ significantly between children with unilateral cleft lip and the controls. Except for second intermolar width, statistically significant differences existed in mean linear maxillary arch dimensions between the unilateral cleft lip and the unilateral cleft lip and palate groups; the mean linear maxillary arch dimensions were significantly greater in the control group than in the unilateral cleft lip and palate group. The mean cleft-affected anterior quadrant length appeared to be the arch dimension with the greatest power of discrimination among the three groups. There were no significant differences in mean linear mandibular arch dimensions among the three groups. CONCLUSIONS: Anterior crossbite was almost three times more common in the unilateral cleft lip and palate group than in the unilateral cleft lip group. Mean linear maxillary arch dimensions differed significantly between the unilateral cleft lip and palate group and the control group. There were no significant differences in mean linear maxillary arch dimensions between unilateral cleft lip and controls or between mean linear mandibular arch dimensions for unilateral cleft lip, unilateral cleft lip and palate, and controls.     

Nasal respiratory resistance in cleft lip and palate

The present study aimed to determine the nasal respiratory resistance (NRR) in subjects with cleft deformity of the lip and palate. The cleft sample was subdivided into cleft lip (N = 15) (mean age 13.4 years, median age 14 years), cleft palate (N = 19) (mean age 12.7 years, median age 12 years), and unilateral cleft lip and palate subjects (N = 27) (mean age 16.4 years, median age 16 years). A control group consisted of 38 patients (mean age 14.4 years, median age 12 years) attending for orthodontic diagnosis. Respiratory resistance was determined by a Mercury rhinomanometer, which was calibrated dynamically before each session. Subjects were given 0.1% xylometazoline hydrochloride half an hour before each session. Resistance was determined unilaterally by the anterior method as well as bilaterally by the posterior method. The results showed that the bilateral nasal resistance did not differ significantly between the cleft samples and the controls. Unilateral measurements of nasal resistance showed higher values for the cleft side than for the noncleft side, both in the cleft lip (CL) and the unilateral cleft lip and palate (UCLP) samples (p less than 0.001). In the cleft palate sample as well as in the controls, unilateral nasal resistance did not differ between the two sides.     

Facial characterization of infants with cleft lip and palate using a three-dimensional capture technique.

OBJECTIVE: To characterize the soft tissue features of infants with unilateral cleft lip (UCL) and unilateral complete cleft lip and palate (UCLP) prior to primary surgery and compare with noncleft controls. DESIGN: Prospective controlled capture of the facial morphology of infants using a noninvasive three-dimensional stereophotogrammetry method. PARTICIPANTS: 23 children with presurgical cleft: 11 UCL (M = 6, F = 5); 12 UCLP (M = 9, F = 3), and 21 noncleft controls (M = 7, F = 14) were imaged at approximately 3 months of age (range 10 to 16 weeks). MAIN OUTCOME MEASURE: Accurate, repeatable quantification of facial soft tissues in infants with clefts prior to surgery. RESULTS: Significant differences (p <.05) were found between the UCLP group and UCL and control groups in anatomical and soft nose width, cleft-side alar wing length, and nasal tip horizontal displacement. Both cleft groups were significantly different from controls and from each other in cleft-side nostril dimensions, alar wing angulation, columella angle, and alar base to corner of mouth dimension; alar base width; and soft tissue defect in nose and the lip and philtrum length bordering the cleft. Significant differences between clefts and controls were identified in the nostril and philtrum on the noncleft side. CONCLUSIONS: The use of children with UCL as controls for UCLP studies is inappropriate. This technique overcame the limitations of direct measurement of infant faces to aid the surgeon in the planning and subsequent re-evaluation of surgical rationale.     

A comparative study of prespeech vocalizations in two groups of toddlers with cleft palate and a noncleft group.

OBJECTIVE: This study examined the prelinguistic contoid (consonant-like) inventories of 14 children with unilateral cleft lip and palate (C-UCLP) at 13 months of age. The children had received primary veloplasty at 7 months of age and closure of the hard palate was performed at 3-5 years. The results of this investigation were compared to results previously reported for 19 children with cleft palate and 19 noncleft children at the age of 13 months. The children with clefts in that study received a two-stage palatal surgery. This surgical procedure was formerly used at our center and included closure of the lip and hard palate at 3 months of age and soft palate closure at 22 months of age. DESIGN: Retrospective study. SETTING: The participants were videorecorded in their homes during play with their mothers. The videotapes were transcribed independently by three trained speech pathologists. PATIENTS: Fourteen consecutive patients born with C-UCLP and no known mental retardation or associated syndromes served as subjects. RESULTS: The children who received delayed closure of the hard palate demonstrated a significantly richer variety of contoids in their prespeech vocalizations than the cleft children in the comparison group. Both groups of subjects with clefts had significantly fewer plosives in their contoid inventory than the noncleft group, and there was no difference regarding place of articulation between the group that received delayed closure of the hard palate and the noncleft group.     

Asian oral-facial cleft birth prevalence.

OBJECTIVE: To determine the clefting birth prevalence among Asian populations, specifically Chinese and Japanese, using raw counts from nonoverlapping published studies of Asian populations, and to investigate whether Asian clefting rates have been interpreted accurately as being up to twice the Caucasian rate. DESIGN: A literature review of articles giving raw counts of clefting in Asian populations, primarily Japanese and Chinese. MAIN OUTCOME MEASURES: Where possible, clefts were identified by the patients' ethnicity, country of origin, cleft type, syndromic status, and birth status. RESULTS: Prevalence rates of cleft lip with or without cleft palate per 1000 live births are reported. Syndromic plus nonsyndromic cleft lip with or without cleft palate: Chinese, 1.30; Japanese, 1.34; Other Asian, 1.47; and total, 1.33. Nonsyndromic cleft lip with or without cleft palate: Chinese, 1.20; Japanese, 1.18; Other Asian, 1.22; and total, 1.19. CONCLUSIONS: Overall, Chinese and Japanese live birth prevalence rates for nonsyndromic cleft lip with or without cleft palate, based on the published reports of birth prevalence, are significantly lower than the oft-quoted rate of 2 per 1000 for Asians. The apparent reason for the discrepancy is that many published prevalence rates included all pregnancies (live births plus pregnancy losses) and do not distinguish between syndromic and nonsyndromic clefts or between cleft palate alone and cleft lip with or without cleft palate. These results demonstrate that it is extremely important for current population-based studies of clefts to include careful delineation of population groups, syndromes, cleft type, and birth status.     

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.     

Closure of cleft lips with Pfeifer's wave line technique does not inhibit upper lip growth: A retrospective study

The aim of this study is to describe the growth of the upper lip after reconstruction with a Pfeifer wave-line incision in patients with unilateral and bilateral cleft lip and palate (CL/P) in the long term.This was a longitudinal, monocentric, retrospective study. Metric standardized lip length measurements were taken annually from the age of 6 months to the age of 16 years. Defined anatomical points were determined which describe the lip length from the nasal entrance to the highest point of the Cupid's bow. The lip length of the unaffected side in unilateral cleft patients was taken as control.A total of 234 patients with cleft lip with/without cleft palate (CL/P) were included in the study. At the time of the primary surgery, the medial sides in unilateral clefts were 2–4 mm and the lateral sides 1.5–2 mm shorter than the normal unaffected side (p≤0.001). Two main periods of growth, one during childhood (first to sixth years) and one during adolescence (12th–16th years) were seen. At the age of 16 years, the end of the observation period, the lip length in unilateral clefts resulted in a clinically not noticeable shortening of the cleft side (0.37±0.26 mm). There was no correlation between lip length development and primary cleft width at the time of primary cleft lip surgery at 6 months. The upper lip in patients with bilateral clefts developed symmetrically without any obvious asymmetry. Both sides showed a lip length difference of 0.1±0.05 mm at the age of 16 years (p=0.1). Compared to the upper lip length of the control group, bilateral clefts showed a slight tendency toward a longer upper lip (p=0.52).Within the limitations of the study it seems that when lip length development is a priority in cleft lip surgery, Pfeifer wave-line procedure is good option to achieve symmetric results in unilateral and bilateral cleft lip surgery and, therefore, is a relevant option among a variety of other techniques.     

The structural implications of a unilateral facial skeletal cleft: a three-dimensional finite element model approach.

OBJECTIVE: For children born with a unilateral facial skeletal cleft, oral motor function is impaired and skeletal development and growth are asymmetrical with regard to the midsagittal plane. This study was designed to verify that a unilateral skeletal cleft and its dimensions (i.e., depth and width) affect the severity of the asymmetric stress and strain distribution within the maxilla. METHODS: A three-dimensional finite element model of a normal maxilla was developed from pediatric, subject-specific computerized tomography scan data. A clefting pattern then was introduced to simulate varying degrees of deformity in geometry, with the bone properties and boundary conditions held constant. The asymmetric index was introduced to quantify the asymmetrical stress and strain distribution within the maxilla with regard to the midsagittal plane. RESULTS: The unilateral skeletal cleft led to a nonuniform, asymmetric stress and strain distribution within the maxilla: intensified on the noncleft side and weakened on the cleft side. As the depth of the unilateral cleft increased, the stress and strain distribution became increasingly asymmetric as measured by the asymmetric index. In contrast, the width of the cleft had minimal effect on the asymmetrical stress and strain distribution. INTERPRETATION/CONCLUSION: These results implied that a child born with a unilateral cleft would be expected to have an asymmetric skeletal development between the noncleft and the cleft sides as a consequence of an asymmetric functional loading pattern.     

Non-syndromic orofacial clefts in Southern Italy: pattern analysis according to gender, history of maternal smoking, folic acid intake and familial diabetes.

BACKGROUND: Genetic studies have demonstrated that non-syndromic clefts of the lip, alveolus and palate have an heterogeneous genetic background, and that environmental factors contribute to the onset of this malformation. Therefore studies on different and homogeneous populations can be useful in detecting potentially related environmental and genetic factors. PURPOSE: The aim of the present study was to evaluate whether gender, folic acid intake, family history of diabetes and/or smoking during pregnancy were associated with a specific type of cleft in a group of patients affected by non-syndromic clefts, collected from Southern Italy. MATERIAL AND METHODS: Data from one hundred and twenty-six patients were evaluated retrospectively. Each cleft was described as composed by separate antomical entities such as lip, alveolus, primary and secondary palate. None had an isolated alveolar cleft and this was used as internal control. Pattern analysis was used to detect differences in the frequencies of any possible combination of 7 types of clefting stratified according to the studied variables. Data were analysed by comparing observed proportions. RESULTS: Isolated cleft palate as well as right-sided clefts of lip, alveolus and palate were more frequent in females (p = 0.0014 and 0.0281, respectively), while left sided clefts were more frequent in males (p = 0.0359). A lack of consumption of folic acid was associated with an higher incidence of clefts of the left lip (p = 0.018), while familial diabetes was associated more often with isolated cleft palate (p = 0.0014). CONCLUSIONS: Gender-related results were comparable with those found in Northern Italy and other countries. Environmentally related results disclosed specific subclasses of clefting associated with lack of folic acid consumption and familial diabetes.