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Background: School absence is associated with lower performance on standardized tests. Children born with orofacial clefts (OFC) are likely to have more absence than children without OFC; however, school absence for children with OFC has not been quantified. We aimed to describe school absence and its relationship with school performance for children with and without OFC., Methods: Population-based record-linked cohort study of children (402 with OFC, 1789 without OFC) enrolled in schools in Western Australia, 2008 to 2012. We compared median school absence rates using Wilcoxon rank tests, and investigated the impact of school absence on standardized scores from reading, numeracy, and writing tests, using multivariable models fitted by generalized estimating equations., Results: In Semester 1, at each primary school year level, children without OFC and children with cleft lip only or cleft palate only had similar median absence rates (approximately 1 week). Children with cleft lip and palate had significantly higher absence rates in Years 4 to 6 (between 1 and 2 weeks). During secondary school, median absence rates were higher (2 weeks) for all children, but not statistically different between children with and without OFC. Higher absence was significantly associated with lower standardized reading, numeracy, and writing scores. However, having a cleft of any type had little influence on the association between absence and test scores., Conclusion: School absence affected school performance for all children. Absence did not differentially disadvantage children born with OFC, suggesting current practices to identify and support children with OFC are minimizing effects of their absence on school performance. Birth Defects Research 109:1048-1056, 2017. © 2017 Wiley Periodicals, Inc., (© 2017 Wiley Periodicals, Inc.)

Background: Educational attainment is important in shaping young people's life prospects. To investigate whether being born with orofacial cleft (OFC) affects school performance, we compared school test results between children born with and without OFC., Methods: Using record-linked datasets, we conducted a population-based cohort study of children liveborn in Western Australia 1980-2010 with a diagnosis of OFC on the Register of Developmental Anomalies, and a random sample of 6603 children born without OFC. We compared odds ratios for meeting national minimum standards in five domains (reading, numeracy, writing, spelling, grammar and punctuation), and adjusted OR (aOR) for children with cleft lip only (CLO), cleft lip and palate (CL + P) and cleft palate only (CPO) for each domain., Results: Results from two testing programs (WALNA and NAPLAN) were available for 3238 (89%) children expected to participate. Most met the national minimum standards. Compared with children without OFC, children with CPO were less likely to meet minimum standards for NAPLAN reading (aOR 0.57 [95%CI 0.34, 0.96]) grammar and punctuation (aOR 0.49 [95%CI 0.32, 0.76]), WALNA writing (aOR 0.66 [95%CI 0.47, 0.92]), and WALNA and NAPLAN numeracy (aOR 0.64 [95%CI 0.43, 0.95] and aOR 0.47 [95%CI 0.28, 0.82]), respectively. Children with CL + P had significantly lower odds for reaching the spelling standard in NAPLAN tests (aOR 0.52 [95%CI 0.29, 0.94]). Children with CLO had similar odds for reaching all minimum standards., Conclusion: Children born with OFC, particularly children with CPO, should be monitored to identify learning difficulties early, to enable intervention to maximize school attainment., (© 2016 John Wiley & Sons Ltd.)

Aim: The aim of this study was to compare hospital admissions from infancy to adulthood, between children born with orofacial clefts (OFC) and those without OFC., Methods: The method used was a cohort study using record-linked administrative datasets. Participants included all children liveborn in Western Australia (WA) between 1980 and 2010 diagnosed with OFC, who were frequency matched by year of birth to randomly selected liveborn children without OFC. We calculated rate ratios (RR) of hospital admission, number and reason of admissions, cumulative length of stay, for each cleft type (cleft lip only (CLO), cleft lip and palate (CL+P), cleft palate only (CPO), no OFC) and by age period (infancy, pre-school, primary and high school ages, and early adulthood)., Results: Overall, 1396 children were diagnosed with an OFC and compared with 6566 children without OFC. Individuals born with OFC were up to three times more likely to be admitted to hospital, had more admissions and longer cumulative length of stay in all age periods. Children with OFC were also more likely to be admitted for ear and digestive system conditions (RR up to 30 and six times higher, respectively). Children with CL+P and CPO were more likely to be admitted for respiratory conditions (RR 1.3-2.0) and children with CPO were six times more likely to be admitted for care for other congenital anomalies., Conclusions: Throughout childhood, individuals born with OFC were more likely to be admitted, and had more hospitalisations than those without OFC. Children born with CL+P or CPO had a higher hospitalisation burden than children born with CLO., (© 2015 The Authors. Journal of Paediatrics and Child Health © 2015 Paediatrics and Child Health Division (Royal Australasian College of Physicians).)

Background: Only two population-based studies have reported survival beyond 15 years for individuals with orofacial clefts (OFC), and only for individuals with isolated OFC. Compared with the general population, long-term survival was similar for individuals with cleft lip only, and lower for individuals with cleft palate only. Results for those born with isolated cleft lip and cleft palate were inconsistent., Methods: Using linked population-based health data, including a congenital anomaly register with active surveillance and diagnoses up to 6 years, we compared survival at 1, 5, and 20 years for infants born 1980 to 2010 with, and without OFC., Results: Of the 8112 live born infants in the cohort, 186 died before 20 years; most (81%) died during infancy. Compared with infants without OFC, infants born with all types of isolated OFC ± additional minor anomalies had similar infant survival (around 99%), but we found lower survival for infants with all cleft types and an additional major anomaly (66-84%). From 1 to 5 years, only infants with cleft palate only and an additional major anomaly had lower survival (97%) compared with children without OFC (99.9%). From 5 to 20 years, children with all cleft types, with or without additional major anomalies had similar survival to children without OFC (98-100%)., Conclusion: Parents with a child diagnosed with an OFC ± additional minor anomalies only can be reassured that the OFC does not influence survival rates in infancy, or long-term. Infant survival was lower only for children with OFC and additional major anomalies., (© 2015 Wiley Periodicals, Inc.)

Background: The teratogenic effects of maternal alcohol consumption during pregnancy include anomalies of craniofacial structures derived from the cranial neural crest cells. The presence of specific craniofacial anomalies contributes to the diagnosis of fetal alcohol spectrum disorders. Cleft lip and palate [orofacial clefts (OFCs)], also derived from the cranial neural crest cells, are common congenital anomalies, but their relationship with prenatal alcohol consumption is unknown., Methods: To evaluate the association between maternal consumption of alcohol during pregnancy and the occurrence of OFCs in infants, we conducted a systematic review and meta-analyses of published studies. We examined the associations between any alcohol consumption, binge level drinking, and heavy and moderate levels of consumption vs. no or low levels of consumption., Results: After screening 737 publications, we identified 33 studies (23 case-control and 10 cohort studies). There was considerable heterogeneity in individual study design, quality measures and study results. Findings from random effects meta-analyses suggest no relationship between prenatal alcohol consumption and the occurrence of OFCs {pooled odds ratios for any alcohol intake and binge level drinking respectively: cleft lip with or without cleft palate 1.00 [95% confidence interval (CI) 0.86, 1.16] from 18,349 participants in 13 studies, 1.04 [95% CI 0.87, 1.24] [8763 individuals, 4 studies]; cleft palate only 1.05 [95% CI 0.92, 1.21] [21,459 individuals, 17 studies], 0.94 [95% CI 0.74, 1.21] [7730 participants, 4 studies]}., Conclusions: While we found no association between alcohol consumption during pregnancy and OFCs in infants, the influence of study design, particularly in relation to alcohol exposure measurement and OFC ascertainment cannot be ignored., (© 2014 John Wiley & Sons Ltd.)

Background: The reported birth prevalence of orofacial clefts (OFCs) varies considerably. This study describes the epidemiology of OFCs in an Australian population., Methods: We studied infants diagnosed with cleft lip, with or without cleft palate (CL±P), and cleft palate only (CPO) since 1980 and reported to the population based Western Australian Register of Developmental Anomalies. We calculated prevalence rates by sex, Aboriginal status, geographic location, and socio-economic status. Associations between clefts and folate availability, pregnancy characteristics, pregnancy outcomes, other congenital anomalies, and age at diagnosis were also investigated., Results: From 1980 to 2009, 917 infants with CL±P (12.05 per 10,000) and from 1980 to 2004, 621 infants with CPO (10.12 per 10,000) were registered. Prevalence rates for CL±P and CPO were 1.9 and 1.3 times higher, respectively, for Aboriginal Australians. Additional anomalies were reported for 31% of infants with CL±P and for 61% with CPO; chromosomal anomalies and other specific diagnoses accounted for 46% and 66%, respectively, of those with CL±P and CPO with additional anomalies. Almost all (99.7%) children with CL±P were diagnosed before 1 year of age, but 12% of CPO diagnoses were made after 1 year of age; 94% of these diagnoses were of submucous clefts and bifid uvula., Conclusions: These data provide a picture of the prevalence of OFCs in WA since 1980, and provide a useful reference for OFC data in Australia and internationally. The quality and completeness of the WARDA data are high, reflected in high prevalence rates, and proportions of clefts occurring with other anomalies., (Copyright © 2012 Wiley Periodicals, Inc.)