An Epidemiologic Study of Oral Clefts in Iran: Analysis of 1669 Cases

2000 ◽  
Vol 37 (2) ◽  
pp. 191-196 ◽  
Author(s):  
Mohammad Hossain Rajabian ◽  
Mehdi Sherkat

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 1669 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 1000 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 Europian than Arab-African or South East Asian countries. However, significant variations in other epidemiologic and some genetic features were observed.

2001 ◽  
Vol 38 (4) ◽  
pp. 358-373 ◽  
Author(s):  
Hans Dotevall ◽  
Hasse Ejnell ◽  
Björn Bake

Objectives: (1) To study the nasal airflow patterns during the velopharyngeal closing phase in speech produced by children with and without cleft palate. (2) To compare the nasal airflow patterns in bilabial, dental, and velar articulation in these children. Design: Prospective, cross-sectional study of a consecutive series of children with cleft palate referred for routine speech evaluation and controls. Setting: Sahlgrenska University Hospital, Göteborg, Sweden. Participants: Seventeen children with cleft lip and palate or cleft palate only and 22 controls aged 7 and 10 years. Method: Nasal airflow was transduced with a pneumotachograph attached to a nose mask and registered together with the acoustic speech signal. Sentences containing nasal-to-stop combinations in bilabial, dental, and velar articulatory positions were used. Main Outcomes Measures: The duration from peak to 5% nasal airflow, the maximum flow declination rate, and the nasal airflow at selected points in time during the transition from nasal-to-stop consonants. Results: In the cleft palate group, duration from peak to 5% nasal airflow was clearly longer than among the controls (p < .0001). The declination of airflow was slower (p < .006) and the rate of nasal airflow at the release of the stop consonant was higher (p < .004) in the cleft palate group. Differences between bilabial versus dental and velar articulation were found in the control group. Conclusion: Studies of the temporal and dynamic characteristics of the nasal airflow variations during speech appear potentially useful for the assessment of velopharyngeal function.


2020 ◽  
Vol 20 (1) ◽  
pp. 257-263
Author(s):  
Adriana Boeri Freire Tamburini ◽  
Ygor Henrique Pereira Rodrigues ◽  
Daniella Reis Barbosa Martelli ◽  
Letízia Monteiro de Barros ◽  
Rodrigo Soares de Andrade ◽  
...  

Abstract Objectives: to investigate the prevalence of dental anomalies in complete deciduous dentition of children with NSCL/P. Methods: this study included 75 children with NSCL/P and 286 healthy control. In both groups the children had deciduous dentition with ages varying from 4 to 6 years. Clinical examination, panoramic and periapical radiographies were performed and dental anomalies of number and shape were considered. Results: there was a higher prevalence of dental anomalies in the case group, compared to the control group. In all, 42 dental anomalies were identified, 25.33% in the case group and 8.04% in control group (p<0.001). Therewas a higher frequency of dental anomalies in NSCL/P (47.36%), followed by non-syndromic cleft lip (31.57%) and non-syndromic cleft palate (21.05%). The occurrence of agenesis (p= 0.005) and twinning (p = 0.029) were higher in the case group. Conclusions: the occurrence of agenesis and dental twinning was more frequent in the case group and may contribute to the definition of oral cleft subphenotype.


2017 ◽  
Vol 54 (4) ◽  
pp. 371-380 ◽  
Author(s):  
Nicola G. Clausen ◽  
Dorthe A. Pedersen ◽  
Jacob K. Pedersen ◽  
Susanne E. Møller ◽  
Dorthe Grosen ◽  
...  

Objective Early life exposure to anesthesia and surgery is suspected to associate with cognitive impairment later in life. We compared academic achievement among adolescents with cleft lip only (CL), cleft palate only (CP), and cleft lip and cleft palate (CLP) with a noncleft control group to investigate whether outcome depends on timing and number of operations during childhood and/or type of oral cleft. Design Nationwide register-based follow-up study. Setting Danish birth cohort 1986 to 1990. Participants Five hundred fifty-eight children with isolated CL (n = 171), CLP (n =222), or CP (n = 195), of which 509 children had been exposed to anesthesia and one or more cleft operation(s), and a 5% sample of the birth cohort (n = 14,677). Main Outcome Measure(s) Test score in the Danish standardized ninth-grade exam and proportion of nonattainment, defined as “results for ninth-grade exam unavailable.” Data adjusted for sex, birth weight, parental age, and parental level of education. Results Compared to controls, children with CL achieved higher scores (mean difference 0.12, 95% CI –0.05; 0.29) and children with CLP presented with lower scores (mean difference –0.06, 95% CI –0.21; 0.09), albeit both statistically insignificant. Children with CP achieved significantly lower scores, mean difference –0.20 (95% CI –0.38; –0.03). Odds ratios for nonattainment at final exam were: CL 0.79 (95% CI 0.46; 1.35), CLP 1.07 (95% CI 0.71; 1.61), CP 2.59 (95% CI 1.78; 3.76). Conclusions Oral cleft type rather than number and timing of anesthesia and operations associate to poorer academic performance. Although a potential neurotoxic effect due to anesthetic agents is not reflected in the data, it cannot be completely excluded.


2005 ◽  
Vol 42 (2) ◽  
pp. 185-191 ◽  
Author(s):  
Diego F. Wyszynski ◽  
Claudia Perandones ◽  
Patricia Yannibelli ◽  
Ricardo D. Bennun

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.


2021 ◽  
pp. 105566562098435
Author(s):  
Lauren L. Madhoun ◽  
Meghan O’Brien ◽  
Adriane L. Baylis

Objective: To examine feeding skills of infants with cleft palate with or without cleft lip (CP±L) using infant-driven feeding systems compared to healthy controls on standard bottles. Design: Cross-sectional cohort. Setting: Large pediatric academic medical center in the Midwestern United States. Participants: Infants with CP±L (n = 15) using the Dr. Brown’s Specialty Feeding System and typically developing infants without CP±L (n = 15) using the Dr. Brown’s Natural Flow Original or Options bottles. Main Outcome Measure(s): Bottle-feeding proficiency, duration, milk transfer, and signs of feeding difficulty. Results: Five-minute feeding proficiency differed significantly between groups with the control group taking 45% of the feed compared to 16% for the CP±L group on level 1 ( P < .001) and 30% on level 2 ( P < .001) nipples. Proportion of milk transfer was 96% ± 7% for controls and 75% ± 24% for the CP±L group ( P = .013). Feeding duration (minutes) differed between the control group (13 ± 3) and the CP±L groups on each nipple level (level 1: 29 ± 16; P = .003; level 2: 32 ± 11; P = .001). Milk transfer rate (mL/min) was 9 ± 3 for control infants compared to 3 ± 1 for infants with CP±L on level 1 ( P < .001) and 5 ± 1 on level 2 ( P = .007). Coughing occurred in 40% of infants with CP±L and 27% of controls. Conclusions: Even when using specialty bottles, infants with CP±L differ from noncleft infants in feeding proficiency, duration, and overall intake.


2019 ◽  
Author(s):  
Najla Alrejaye ◽  
Jonathan Gao ◽  
Snehlata Oberoi

AbstractIntroductionThe aim of this study was to evaluate three dimensionally the effect of the combined maxillary expansion and protraction treatment on oropharyngeal airway in children with non-syndromic cleft palate with or without cleft lip (CP/L).MethodsCBCT data of 18 preadolescent individuals (ages, 8.4 ± 1.7 years) with CP/L, who underwent Phase I orthodontic maxillary expansion with protraction, were compared before and after treatment. The average length of treatment was 24.1± 7.6 months. The airway volume and minimal cross-sectional area (MCA) were determined using 3DMD Vultus imaging software with cross-sectional areas calculated for each 2-mm over the entire length of the airway. A control group of 9 preadolescent individuals (ages, 8.7 ± 2.6 years) with CP/L was used for comparison.ResultsThere was a statistically significant increase in pharyngeal airway volume after phase I orthodontic treatment in both groups, however, there was no statistically significant change in minimal cross-sectional area in neither study nor control group.ConclusionThe findings showed that maxillary expansion and protraction did not have a significant effect on increasing oropharyngeal volume and MCA in patients with CP/L.


2006 ◽  
Vol 43 (5) ◽  
pp. 571-576 ◽  
Author(s):  
Linda D. Vallino-Napoli ◽  
Merilyn M. Riley ◽  
Jane L. Halliday

Objective: To describe the epidemiological characteristics of oral clefts occurring with other birth defects in Victoria, Australia. Methods: Information on infants and fetuses reported to the Victorian Birth Defects Register from 1983 to 2000 was collected. Birth defects were classified as Pierre Robin Sequence, chromosomal anomaly, nonchromosomal syndrome, single-system defect, or multiple-system defect. Pregnancy outcome and associations with selected infant and maternal features was examined. Results: One third of the 2022 oral clefts recorded had other birth defects. There were more overall cleft cases involving multiple systems and chromosomal anomalies than any other birth defect group. The prevalence ratio of cleft lip with or without cleft palate (CL/P) was highest among multiple-system defects and chromosomal anomalies. Perinatal mortality was high, with termination of pregnancy highest in CL/P and chromosomal anomalies and multiple-system defects. There was a nonsignificant excess of clefts among multiple births. Women ≥40 years old had a tendency toward having a child with a cleft palate and another birth defect. There was an increased likelihood that women born in the U.K. would have a baby with CL/P and another birth defect. Conclusions: This is among the first reports in Victoria, Australia, describing oral clefts and other birth defects and associations between infant and maternal factors. Although some findings confirmed other population-based studies, some continued to be at variance. Nonetheless, the data derived support examination of babies diagnosed with clefts for associated comorbidities.


2021 ◽  
pp. 105566562110132
Author(s):  
Justin Parry Weinfeld ◽  
Jakob Åsberg Johnels ◽  
Christina Persson

Objective: To investigate the prevalence of reading difficulties in children born with cleft palate at ages 9 and 10 in Sweden. Design: Using a cross-sectional design, a parental questionnaire assessing dyslexia-like reading difficulties (Short Dyslexia Scale, SDS) was administered together with separate questions regarding background data. Participants: Families with a child born with overt cleft palate with or without cleft lip in 4 regions of Sweden. A total of 245 families were approached of which 138 families responded. Data from 136 (56%) were complete with information on cleft type and could be analyzed. Results: Twenty-two percent (95% CI, 15-30) of the whole study group displayed risk for dyslexic reading difficulties on the SDS corresponding to the 7th to 10th percentiles in the population. Children with cleft palate only had a significantly higher prevalence of reading difficulties (37%) compared to children with unilateral cleft palate (19%) and bilateral cleft palate (10%). The frequency of reading difficulties in participants with comorbidity was 32%. Among a subgroup with reported comorbidity in areas of attention, language, and learning problems, there was a 2.5 times higher risk of reading disability compared to participants without this reported comorbidity. Conclusion: The prevalence of reading difficulties in the cleft palate population was higher than in the general population. Results showed that co-occurring difficulties were common in the cleft group and that reading difficulties often appear together with other, co-occurring neurodevelopmental difficulties.


2020 ◽  
pp. 105566562098024
Author(s):  
Kim Bettens ◽  
Laura Bruneel ◽  
Cassandra Alighieri ◽  
Daniel Sseremba ◽  
Duncan Musasizib ◽  
...  

Objective: To provide speech outcomes of English-speaking Ugandan patients with a cleft palate with or without cleft lip (CP±L). Design: Prospective case–control study. Setting: Referral hospital for patients with cleft lip and palate in Uganda. Participants: Twenty-four English-speaking Ugandan children with a CP±L (15 boys, 9 girls, mean 8.4 years) who received palatal closure prior to 6 months of age and an age- and gender-matched control group of Ugandan children without cleft palate. Interventions: Comparison of speech outcomes of the patient and control group. Main Outcome Measures: Perceptual speech outcomes including articulation, resonance, speech understandability and acceptability, and velopharyngeal composite score (VPC-sum). Information regarding speech therapy, fistula rate, and secondary surgery. Results: Normal speech understandability was observed in 42% of the patients, and 38% were judged with normal speech acceptability. Only 16% showed compensatory articulation. Acceptable resonance was found in 71%, and 75% of the patients were judged perceptually to present with competent velopharyngeal function based on the VPC-sum. Additional speech intervention was recommended in 25% of the patients. Statistically significant differences for all these variables were still observed with the control children ( P < .05). Conclusions: Overall, acceptable speech outcomes were found after early primary palatal closure. Comparable or even better results were found in comparison with international benchmarks, especially regarding the presence of compensatory articulation. Whether this approach is transferable to Western countries is the subject for further research.


2020 ◽  
Vol 57 (6) ◽  
pp. 671-677 ◽  
Author(s):  
Yah-Huei Wu-Chou ◽  
Kuo-Ting Philip Chen ◽  
Yi-Chieh Lu ◽  
Yin-Ting Lin ◽  
Hsien-Fang Chang ◽  
...  

Objective: Nonsyndromic oral clefts are common birth defect with complex etiology. In the present study, we attempt to further validate the possible role for ABCA4 and ARHGAP29 in the susceptibility to nonsyndromic oral clefts. Design: We performed allelic transmission disequilibrium test analysis, on 10 eligible single nucleotide polymorphisms (SNPs) and SNP haplotypes using the Family-Based Association Test. Participants: The study sample consisted of 334 case–parent trios of nonsyndromic oral clefts from Taiwanese population, separated into nonsyndromic cleft lip with or without cleft palate (NSCL/P) and nonsyndromic cleft palate only (NSCPO) groups. Results: We found only the SNP rs560426 within the ABCA4 gene showed strong association with NSCPO ( P = .03498; Permuted P = .05382). No association between other 9 selected SNPs in ABCA4-ARHGAP29 region and the risk of nonsyndromic oral clefts was found. For the haplotype analyses, we found only haplotype T-C (rs570926 and rs3789431) in ABCA4 block 2 showed significant association with nonsyndromic NSCL/P in these Taiwanese trios. Conclusions: We used a family-based analysis in 334 Taiwanese case–parent trios to validate the possible role for ABCA4 and ARHGAP29 in the susceptibility to nonsyndromic oral clefts. This study provides a new evidence for an association between the intron variant rs560426 within ABCA4 and nonsyndromic cleft palate which may contribute their regulatory role in craniofacial development.


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