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PubMed1977

The influence of primary unilateral cleft lip repair on facila growth.

The lip pressure exerted by the repaired cleft lip was studied in 35 rabbits during a 20-week period. Animals were divided into four groups. Two of them were controls, and in the other two, two different surgical procedures were used for the lip repair. The results of this study indicate that there was reduction in lip pressure resulting from the surgically induced cleft lip, alveolus, and palate. Substantial increases in lip pressure were shown in both groups in which lip repair was completed. The influence of primary unilateral cleft lip repair on the facial growth of rabbits in this study will be presented in Part II of this report.

Animals↗
PubMed1998

Comparative study of diagnostic measures in borderline surgical cases of unilateral cleft lip and palate and noncleft Class III malocclusions.

Patients with complete unilateral cleft lip and palate present difficult growth problems. Their anteroposterior discrepancies in jaw and dentition are frequently so severe that some epidemiologic studies report the necessity of orthognathic surgery in 25% of their sample. The aims of this study were three-fold: (1) to delineate diagnostic measures in borderline surgical cases of unilateral cleft lip and palate, (2) to verify the significance of negative overjet as a measure of anteroposterior discrepancy, and (3) to compare these diagnostic measures with those of borderline surgical cases of noncleft Class III malocclusions. The sample consisted of 29 patients with unilateral cleft lip and palate and 25 noncleft Class III Korean patients (mean age, 18.69 years); all had crossbites of all four incisors. Each of their pretreatment study casts and cephalograms were analyzed. The group with unilateral cleft lip and palate was divided into two subgroups on the basis of the method of their anterior crossbite resolution; 18 subjects were treated with orthodontics alone (Cleft-NS) and 11 subjects with orthognathic surgery (Cleft-Surg). The noncleft Class III group was divided into two subgroups; 6 of the subjects were orthodontically treated (Cl III-NS), and 19 were surgically treated (Cl III-Surg). The group with unilateral cleft lip and palate showed smaller SNA and SNB angles than the noncleft Class III group, but the ANB angles and the amount of anterior crossbites showed no statistical differences. When the Cleft-NS and the Cleft-Surg groups were compared, the ANB angle and the Wits measurements were significantly different. When the Cl III-NS and Cl III-Surg groups were compared, the SNB, ANB, L1GoGn, Wits, and the crossbite showed significant differences. For borderline surgical Class III unilateral cleft lip and palate cases, ANB angle, Wits appraisal, and ABGoGn angle were critical diagnostic parameters. On the other hand, the magnitude of anterior crossbite, the negative overjet, was shown not to be a significant measure of anteroposterior discrepancy.

Adolescent↗
PubMed1992

Reproductive history of mothers of children with solitary, nonsyndromic cleft lip and/or palate.

The reproductive history was studied to evaluate if the three types of solitary, nonsyndromic clefts: cleft lip (CL), cleft lip and palate (CLP), and cleft palate (CP) are homogeneous entities. Occurrence of fetal loss, maternal health, and drug consumption of the mother during the pregnancy were compared in cases involving three types of clefts. Data was gathered from 87 children with clefts, 55 males and 32 females. Spontaneous abortions and vaginal bleeding were found to occur significantly more often in the (older) mother of a CLP child. This suggests that the factors involved in the etiology of CLP differ from the factors involved in CL and CP. Therefore, grouping of data of the three types of clefts in studies on the etiology should be avoided.

Abortion, Spontaneous↗
PubMed2004

Transforming growth factor-alpha and nonsyndromic cleft lip with or without palate in Brazilian patients: results of a large case-control study.

OBJECTIVE: Transforming growth factor-alpha (TGFA) was the first gene suggested to be associated with nonsyndromic cleft lip, cleft palate, or both (CL/ P). There are, however, still controversies of the effect of TGFA on the predisposition of this malformation. To contribute to a better understanding of the role of this gene in the occurrence of CL/P we undertook a case-control study including patients and controls ascertained in different regions of the country. DESIGN: We examined the C2/TaqI variant of the TGFA gene in 536 patients with nonsyndromic CL/P and 412 controls. The TGFA genotype frequencies in patients were compared with controls using chi-square or Fisher exact test. DNA, obtained from peripheral blood or buccal swabs, was genotyped for the TaqI polymorphism of TGFA. SETTING: The probands and corresponding controls were ascertained in different centers of Brazil, partly representing the ethnic admixture of our population. RESULTS: The TGFA genotype distribution was very similar in patients with CL/P ascertained in the three different regions of Brazil. However, a discrete difference was observed between controls of Säo Paulo and Ceará (chi-square = 3.605; p = 0.058), with a lower value of the C2/Taq allele frequency in controls of CE (0.04). These data reinforce that this polymorphic system is heterogenous among different ethnic groups. In addition, no evidence was found for an association of TGFA with CL/P in this case-control study. CONCLUSION: These data further suggest that TGFA is not a relevant modifier locus for the occurrence of CL/P.

Adolescent↗
PubMed2001

Failure to thrive in babies with cleft lip and palate.

We established the frequency of failure to thrive (FTT) in children undergoing primary cleft procedures by using growth charts and standard-deviation scores. Initially, 147 babies with cleft lip and/or palate undergoing 186 primary lip-and-palate repairs were studied between 1993 and 1996. Rates of FTT were categorised according to cleft type. There was an increasing rate of FTT from 32% for unilateral cleft lip and palate to 38% for bilateral cleft lip and palate to 49% for cleft palate. There was a high incidence of FTT in palatal clefts, especially if these were associated with a syndrome or anomaly (P= 0.001). The incidence of FTT with the Pierre Robin sequence was 100%. In view of the high rates of FTT, two changes were instituted: a feeding-support nurse was appointed to supervise and monitor patients at risk and all patients with the Pierre Robin sequence had supervised airway management. Thereafter, the incidence of FTT was prospectively studied in 68 babies undergoing 84 primary procedures between 1997 and 1999. There was a decrease in the incidence of FTT in comparison with the earlier cohort (9% for unilateral cleft lip and palate, 20% for bilateral cleft lip and palate, 26% for cleft palate). There was a significant decrease in the incidence of FTT in the group with the Pierre Robin sequence, from 100% to 40%. As a result of the provision of a feeding-support nurse and airway management of patients with the Pierre Robin sequence, the incidence of FTT was reduced and the audit loop closed.

Cleft Lip↗
PubMed2004

Cleft lip and palate in Jordan: birth prevalence rate.

OBJECTIVE AND DESIGN: A retrospective investigation was conducted to determine the prevalence rate of Jordanian children born with oral clefts from 1991 to 2001. SETTING: Hospital surgical records from two main sources were used to identify all children born with orofacial clefting. Information about date of birth, sex, cleft types, and associated major anomalies and syndromes were recorded. RESULTS: The overall prevalence rate for live births with cleft lip, cleft palate, or both was 1.39 per 1000 live births. Thirty percent of the clefts identified affected the lip, 22 percent affected the palate, and 48 percent involved the clefts of the lip and palate. In general, higher prevalence rates were found for boys than girls (55% boys versus 45% girls). There was a statistically significant sex difference between the cleft types. Clefts of the isolated palate, however, were more common in girls. Eighteen percent of patients with cleft were associated with major anomalies or syndromes. CONCLUSIONS: The reported prevalence rate for orofacial clefts among Jordanians is similar to the previously reported prevalence rate in white Caucasians. Results and future recommendations are discussed.

Arabs↗
PubMed2003

Development of a network system for the care of patients with cleft lip and palate in Thailand.

Cleft lip and palate are major public health concerns in Thailand, particularly in the north-east. The challenge for a developing country is to adapt to its unique environment the form of multidisciplinary care of patients with clefts in industrialised nations. An epidemiological study of the incidence of clefts was made by reviewing previous studies conducted in Thailand as a guide to development of a prospective multicentre study. As a result, a system for the care of patients with clefts has been developed by the Cleft Center in Khon Kaen University. The new aims of cleft care in Thailand are to establish and develop specialised cleft centres and supportive systems for interdisciplinary management, adopt protocols for long-term guidelines, provide education programmes about clefts, develop a database, and conduct relevant research projects. Other key factors in its success are the multicentre project, collaboration, fund raising, and good public relations.

Cleft Lip↗
PubMed2000

Maxillary sinus development and sinusitis in patients with cleft lip and palate.

OBJECTIVE: Sinusitis is frequently associated with cleft lip and palate. The present study was performed in an attempt to investigate maxillary sinus development in relation to the occurrence of sinusitis in cleft patients. METHODS: Forty-seven consecutive patients with cleft lip and alveolus with or without cleft palate underwent computed tomographic scans of the maxilla. The cross-sectional area of the maxillary sinus at the level of the zygomatic arch was measured, and the soft tissue density shadow of the sinus was scored on a 4-point scale. RESULTS: The cross-sectional area of the sinus was significantly smaller in the child group (10 years or less) than in the adolescent group (11-20 years, P<0.001) and the adult group (over 20 years, P<0.02). The cross-sectional area significantly increased with age in the child group (r=0.552, P<0.001). The growth rate became slower in the adolescent group. The cross-sectional area then gradually decreased with age in the adult group (r=-0.64, P<0.05). Sinusitis was observed in 15 patients (32%) and in 20 sinuses (21%). Severe sinusitis was more common in the child group than in the patients over 10 years of age (P<0.05). CONCLUSION: These results indicate that maxillary sinusitis associated with cleft lip and palate occurs preferably in the developing sinuses of children.

Adolescent↗
PubMed2006

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.

Adolescent↗
PubMed1984

Left cleft lip predominance and genetic similarities of L line and CL/Fr strain mice.

Newborn litters of the L line and CL/Fr and A/JFr strains were examined, and sex, frequency and type of cleft lip (left, right or bilateral) were recorded. Embryos and fetuses from crosses between these strains and line were collected on days 13 to 16 of gestation, and frequency and type of cleft lip recorded. Overall cleft frequencies in L X CL/Fr, CL/Fr X L, and CL/Fr X A/JFr crosses (female stated first) were similar, while in A/JFr X L (10.3%) they were significantly lower than in L X A/JFr (23.3%). The data suggested that the same maternal effect genes were present in CL/Fr and the related L line and absent from A/JFr. In the L, CL/Fr, and A/JFr newborns, there was a tendency for males to have higher frequencies of cleft lip and bilateral cleft lip and the latter was significant for L. Left cleft lip frequency was significantly higher than right for L and CL/Fr newborns and in embryos of the CL/Fr X L and L X CL/Fr cross. No significant differences in laterality were found in the A/JFr strain, A/JFr X L, L X A/JFr, and CL/Fr X A/JFr crosses. It was concluded that (1) the embryonic and maternal effect genes for cleft lip are similar or identical in CL/Fr and L; and (2) using data from the literature, there are additional genetic factor(s) increasing left cleft lip occurrence acting in the embryo, which are present in CL/Fr, L, A/HeJ, A/He, and A/St and absent from A/JKt, A/J, A/JFr, and A/WySn.

Animals↗
PubMed2005

Cross-sectional study of the length of the nasal bone in cleft lip and palate subjects.

OBJECTIVE: The purpose of this study was to analyze the human nasal bone length in newborns and male adults with cleft lip (CL), cleft palate (CP), and unilateral cleft lip and palate (UCLP) and to compare the results to previous findings in prenatal material. DESIGN: This study was a radiographic profile cephalometric cross-sectional analysis. SUBJECTS AND METHODS: Profile radiographs from 60 newborns with a male-to-female ratio of 1:1 in each group (20 CL, 20 CP, and 20 UCLP) and 60 male adults (20 CL, 20 CP, and 20 UCLP) were randomly selected among radiographs, taken for optimizing the treatment planning. The nasal bone lengths (n-na) were measured with a digital caliper on the profile radiographs. To compare the nasal bone lengths in the different cleft groups, Student's t tests at a significant level at 5% were performed. RESULTS: Nasal bone length was significantly shorter in male adult patients with CL compared with patients with CP. Furthermore, the nasal bone length was significantly shorter in newborns with CL (2 months) compared to newborns with UCLP (2 months). A borderline significance was seen in the comparison of patients with CL and UCLP in male adults. There was no significant difference in the nasal bone length between the patients with CP and UCLP, in either the newborns or the male adults. CONCLUSIONS: Nasal bone length was significantly shorter in subjects with CL compared with subjects in whom the palate was clefted. The results show that the clefted lip in CL is associated with a subjacent skeletal deviation in the upper midface.

Adult↗
PubMed2001

The effects of lactation education and a prosthetic obturator appliance on feeding efficiency in infants with cleft lip and palate.

OBJECTIVE: This prospective study examined the effect of lactation instruction and palatal obturation in decreasing time to feed, increasing intake, and on growth in eight breast milk bottle-fed newborn infants with cleft lip, cleft palate, or both. DESIGN: An A, B1, C1, B2, and C2 reversal design was used with eight mothers. In A, baseline data on minutes fed by breast were recorded. In B1, baseline on minutes fed with a Haberman bottle was recorded. In C1, minutes fed following lactation education and palatal obturation were documented. Lactation education was information given to mothers to recognize infant feeding cues and to have infant-led feedings. The palatal obturator was a passive molding appliance. In B2, the obturator was removed and minutes fed noted. In C2, the obturator was returned and lactation support provided. Mothers kept feeding logs, satisfaction was assessed, and infant breast milk intake and flow rate were recorded during each study phase. Routine nutrition evaluation of weight, height, weight for height, and feed volume was completed by a registered dietitian during and following completion of the study. RESULTS: Feeding times decreased with all infants, volume consumed increased with seven of eight infants, and flow rate increased with all infants. Mean feeding times during B1 and B2 phases (Haberman bottle only) were 34.4 and 32.3 minutes, respectively. Mean feeding times during C1 and C2 phases (obturation and lactation education) were 15.1 and 15.6 minutes. Volume of milk consumed during B1 and B2 feedings averaged 36.5 and 37 mL, compared with 67 mL and 76 mL during C1 and C2 phases. Growth as measured by height, weight gain, and weight for height during the study and the first 2 years of life compared favorably with that of children born without clefts. CONCLUSIONS: The combined use of a palatal obturator and lactation education reduced feeding time and increased volume intake and was associated with good growth. Mothers who had desired to breast-feed elected to use the obturator to support high-volume intake, decrease infant fatigue, and provide breast milk for nutrition.

Analysis of Variance↗
PubMed2001

[Incidence of the cleft lip and palate in the University of Chile Maternity Hospital and in maternity Chilean participating in the Latin American Collaborative Study of Congenital Malformations (ECLAMC)].

BACKGROUND: ECLAMC is a registry, aimed to assess the incidence of congenital malformations, that started in 1967 and Chile incorporated to it in 1969. AIM: To report the incidence of cleft lip/palate, updated to 1999 in the University of Chile Maternity Hospital and other Chilean hospitals participating in the ECLAMC. PATIENTS AND METHODS: A review of the ECLAMC database that registers all births or stillbirths of more than 500 g. RESULTS: The incidence of orofacial cleft, at the University of Chile Maternity Hospital, in the period 1991-1999 was 17.8 per 10,000 (12.6 for cleft lip and 5.2 for cleft palate). The incidence in the rest of participating hospitals was 12.04 and 4.6 respectively. Males had a higher incidence of cleft lip and 80% of children with cleft palate, had other malformations, most of them as part of a syndrome (13 and 18 trisomy, holoproscencephalia, Pierre Robin, Apert en EE syndromes, anencephaly etc). In three of 12 children with cleft lip but without cleft palate, there was a relative with the same malformation. CONCLUSIONS: It is proposed that both entities, cleft lip with or without cleft palate and cleft palate without cleft lip, are two etiopathogenically different conditions.

Chi-Square Distribution↗
PubMed2000

Analysis of the candidate genes responsible for non-syndromic cleft lip and palate in Japanese people.

In order to assess the association of alleles for candidate genes with non-syndromic cleft lip and palate, DNA samples from 43 Japanese patients were compared with those from 73 control subjects with respect to the genes encoding transforming growth factor alpha (TGFalpha), TGFbeta and gamma-aminobutyric acid type A receptor beta3 (GABRB3). The restriction fragment length polymorphisms of the 3'-non-coding region of the TGFalpha gene K-primer region were observed after digestion with NcoI and HinfI. Allele 4 was the most common among cases of cleft lip with or without cleft palate, whereas allele 2 was the most common among controls. A significant difference was found in this region between groups with cleft lip (with or without cleft palate) and controls (chi2=10.190; P=0.017). Three alleles of the TGFbeta2 gene were tested, and allele 2 was the most common in both cases and controls. The proportion of allele 2 in the case group was greater than that in the control group, showing a significant difference between cases of cleft lip (with or without cleft palate) and controls (chi(2)=19.208; P<0.0001). No significant differences in variants of TGFbeta3 or GABRB3 between case and control populations were observed. Thus it is concluded that TGF genes play a role in craniofacial development, and that alleles of TGFalpha or/and TGFbeta2 are associated with cleft lip and cleft palate in Japanese populations.

Adolescent↗
PubMed1997

Sagittal position of the left and right maxillary segment in children with cleft lip and palate.

OBJECTIVE: The purpose of the present study was to investigate left-right differences in the sagittal position of the maxillary segments in children with cleft lip and palate. METHOD: The sample consisted of children with operated cleft lip or cleft lip and alveolus [CL/CLA (n = 16) mean age, 9.3 yr], operated unilateral cleft lip and palate [UCLP (n = 27) mean age, 9.1 yr], and operated bilateral cleft lip and palate [BCLP (n = 17) mean age, 9.5 yr]. Computed tomography (CT) horizontal slices of the maxilla were obtained and used to determine the sagittal position of the left and right segment of the maxilla in relation to the mandibular rami and the cranial base. Significant effects were analyzed with multivariate analyses of variance (MANOVA). RESULTS AND CONCLUSIONS: It was concluded that, in contrast to children having CL/CLA or UCLP, children with BCLP showed left-right differences in the sagittal position of the maxillary segments. The segment on the left side was more posteriorly positioned compared to the right side. Because the same results were obtained in relation to the mandibular rami as well as in relation to the cranial base, it can be assumed that the position of these rami are not affected by the different types of oral clefts.

Alveolar Process↗
PubMed1983

Genetic linkage studies with cleft lip and palate: report of two family studies.

Genetic linkage studies are reported on two families with cleft lip +/- cleft palate. For the first family (LP01) the etiology of the clefting is unknown, and the linkage analyses were done assuming both autosomal dominant and autosomal recessive inheritance. Close linkage is rejected with the Duffy blood group under the dominant model and with four loci (Duffy, Kidd, and ABO blood groups and haptoglobin) under the recessive model. The second family (LP02) is a Mexican-American family segregating the van der Woude syndrome with lip pits. The linkage analyses for this autosomal dominant trait excluded close linkage with seven genetic markers, including three on chromosome one. The maximum lod scores were 0.6 with BF (chromosome 6) and 0.4 with the P blood group, which is not yet mapped.

Cleft Lip↗
PubMed1999

Computer-assisted anthropometry for outcome assessment of cleft lip.

Anthropometry and clinical examination best evaluate the morphology of repaired cleft lip and nose. An original, accurate, and practical image analysis of the lip and nose, which takes advantage of the mathematic, geometric, and organizational capabilities of public domain NIH-Image software (http://rsb.info.nih.gov/nih-image/), has been developed and tested over the past 6 years. A modified structured physical examination form that complements this analysis is under study. Accuracy of NIH-Image-based anthropometry was compared with direct measurements of 22 linear distances on the lip and nose. Twenty-five sets of direct measurements were taken, prospectively, on 15 children with repaired cleft lip over a 6-year period. The results were submitted to regression analysis. Then, relevant lip and nasal tip aesthetics were evaluated by the measuring capabilities of NIH-Image to create a quantitative assessment tool. For each episode, 15 possible faults were weighted, according to aesthetics and deformity, to provide an adverse score. The sum of the 5 lip scores, 10 nose scores, and combination gave respective grades. The analysis was modified to stratify congenital deformity to relate severity of disease to outcome. This analysis was applied to digitized images of 19 consecutive children, immediately prior to repair of complete unilateral cleft lip and nose, at the time of palate repair, and annually from the age of 3 to 6 years. There were 19 NIH-Image-based measurements of the congenital deformity and 35 measurements of surgical results; four children had three sets of records, eight had two sets, and seven had one set Descriptive statistics were applied. Following 556 paired direct and computer-assisted measurements, exceptional linear correlation was shown with a Pearson R coefficient of 0.96. The best correlation was lines within the plane of the camera lens, with the average difference ranging between 0.025 and 0.997 mm. Visual inspection of frontal and submental photographs of excellent, good, and poor results substantiates the ability of this analysis to quantify and grade a spectrum of relevant cleft lip and nasal anatomy. For these 19 patients, there was a broad range of performance scores, approximating a normal distribution. The mean of the NIH-Image-based analysis scores, 16.91, was a (very) good grade. A single standard deviation of 6.88 extended up into excellent and down to fair. The congenital analysis indicated a range of deformity. Comparing deformity with outcome, simple regression analysis had a coefficient of determination (R2) of 0.223, indicative of a weak positive relationship. An accurate and practical morphologic computer-assisted outcome assessment of repaired cleft lip and nasal deformity has been developed. There is a weak direct correlation between severity of deformity and outcome. Testing in multiple clinics is warranted.

Anthropometry↗