Risk of Oral Clefts in Twins
- 1 May 2011
- journal article
- research article
- Published by Ovid Technologies (Wolters Kluwer Health) in Epidemiology
- Vol. 22 (3), 313-319
- https://doi.org/10.1097/ede.0b013e3182125f9c
Abstract
Background: Small studies have indicated that twinning increases the risk of oral cleft. Methods: We used data from a Danish national population-based cohort study to investigate whether twinning was associated with isolated oral cleft, and to estimate the twin probandwise concordance rate and heritability. Twins (207 affected/130,710) and singletons (7766 affected/4,798,526) born from 1936 through 2004 in Denmark were ascertained by linkage among the Danish Facial Cleft Database, the Danish Twin Registry, and the Civil Registration System. We computed oral cleft prevalence and prevalence proportion ratio for twins versus singletons, stratified for 3 subphenotypes. Probandwise concordance rates and heritability for twins were estimated for 2 phenotypes—cleft lip with or without cleft palate (CL/P) and cleft palate (CP). Results: The prevalence of oral cleft was 15.8 per 10,000 twins and 16.6 per 10,000 singletons (prevalence proportion ratio = 0.95; 95% confidence interval = 0.83–1.1). This prevalence was similar for monozygotic and dizygotic twins. The probandwise concordance rate was higher for CL/P for monozygotic twins than for dizygotic twins (50% vs. 8%, respectively). A similar contrast was present for CP. Recurrence risk for both types of clefts was greater in dizygotic twins than in non-twin siblings. Heritability estimates were above 90% for both CL/P and CP. Conclusions: No excess risk of oral cleft could be demonstrated for twins compared with singletons. The concordance rates and heritability estimates for both types of clefts show a strong genetic component.Keywords
This publication has 42 references indexed in Scilit:
- Search for Genomic Alterations in Monozygotic Twins Discordant for Cleft Lip and/or PalateTwin Research and Human Genetics, 2009
- A cohort study of recurrence patterns among more than 54 000 relatives of oral cleft cases in Denmark: support for the multifactorial threshold model of inheritanceJournal of Medical Genetics, 2009
- X-chromosome inactivation patterns in monozygotic twins and sib pairs discordant for nonsyndromic cleft lip and/or palateAmerican Journal of Medical Genetics Part A, 2007
- Cleft Lip and Palate versus Cleft Lip Only: Are They Distinct Defects?American Journal of Epidemiology, 2005
- Changing Lifestyles and Oral Clefts Occurrence in DenmarkThe Cleft Palate Craniofacial Journal, 2005
- Discordant MZ Twins With Cleft Lip and Palate: A Model for Identifying Genes in Complex TraitsTwin Research and Human Genetics, 2005
- Multiplex Relative Risk and Estimation of the Number of Loci Underlying an Inherited DiseaseAmerican Journal of Human Genetics, 2002
- The 20th Century Danish Facial Cleft Population—Epidemiological and Genetic-Epidemiological StudiesThe Cleft Palate Craniofacial Journal, 1999
- Cleft lip (± cleft palate) in Danish twins, 1970-1990American Journal of Medical Genetics, 1993
- Congenital malformations in twins in England and Wales.Journal of Epidemiology and Community Health, 1991