Recurrence Risk for Offspring of Twins Discordant for Oral Cleft - A Population-based Cohort Study of the Danish 1936–2004 Cleft Twin Cohort

Dorthe Grosen; Camilla Bille; Jacob Krabbe Pedersen; Axel Skytthe; Jeffrey C Murray; Kaare Christensen

doi:10.1002/ajmg.a.33608

Back

Recurrence Risk for Offspring of Twins Discordant for Oral Cleft - A Population-based Cohort Study of the Danish 1936–2004 Cleft Twin Cohort

Journal article

Open access

Peer reviewed

Recurrence Risk for Offspring of Twins Discordant for Oral Cleft - A Population-based Cohort Study of the Danish 1936–2004 Cleft Twin Cohort

Dorthe Grosen, Camilla Bille, Jacob Krabbe Pedersen, Axel Skytthe, Jeffrey C Murray and Kaare Christensen

American journal of medical genetics. Part A, Vol.152A(10), pp.2468-2474

10/2010

DOI: 10.1002/ajmg.a.33608

PMCID: PMC2946514

PMID: 20799319

Files and links (1)

url

https://www.ncbi.nlm.nih.gov/pmc/articles/2946514View

Open Access

Abstract

Our objective in this Danish population-based cohort study was to estimate the recurrence risk of isolated oral cleft (OC) for offspring of the unaffected co-twins of OC discordant twin pairs and to compare this risk to the recurrence risk in the offspring of the affected co-twin as well as to the risk in the background population. During 1936–2004, 207 twin pairs were ascertained, among whom at least one twin had an OC. The index persons were twins discordant for OC who had children (N=117), and their offspring (N=239). The participants were ascertained by linkage between The Danish Facial Cleft Database, The Danish Twin Registry and The Danish Civil Registration System. In the study OC recurrence risk for offspring of the affected and unaffected twin and relative risk were compared to the background prevalence. We found that among 110 children of the 54 OC affected twins, two (1.8%) children had OC corresponding to a significantly increased relative risk (RR = 10; 95% CI 1.2 to 35) when compared to the frequency in the background population. Among the 129 children of the 63 unaffected twins, three (2.3%) children were affected, corresponding to a significantly increased relative risk (RR = 13; 95% CI 2.6 to 36) when compared the background prevalence. We concluded that in OC discordant twin pairs similar increased recurrence risks were found among offspring of both OC affected and OC unaffected twins. This provides further evidence for a genetic component in cleft etiology and is useful information for genetic counseling of twin pairs discordant for clefting.

Genetics

twins

cleft lip and palate

oral cleft

recurrence risk

Details

Title: Subtitle: Recurrence Risk for Offspring of Twins Discordant for Oral Cleft - A Population-based Cohort Study of the Danish 1936–2004 Cleft Twin Cohort
Creators: Dorthe Grosen - Epidemiology, Institute of Public Health, University of Southern Denmark, Odense, Denmark
Camilla Bille - Epidemiology, Institute of Public Health, University of Southern Denmark, Odense, Denmark
Jacob Krabbe Pedersen - Epidemiology, Institute of Public Health, University of Southern Denmark, Odense, Denmark
Axel Skytthe - Epidemiology, Institute of Public Health, University of Southern Denmark, Odense, Denmark
Jeffrey C Murray - Epidemiology, Institute of Public Health, University of Southern Denmark, Odense, Denmark
Kaare Christensen - Epidemiology, Institute of Public Health, University of Southern Denmark, Odense, Denmark
Resource Type: Journal article
Publication Details: American journal of medical genetics. Part A, Vol.152A(10), pp.2468-2474
DOI: 10.1002/ajmg.a.33608
PMID: 20799319
PMCID: PMC2946514
NLM abbreviation: Am J Med Genet A
ISSN: 1552-4825
eISSN: 1552-4833
Language: English
Date published: 10/2010
Academic Unit: Anatomy and Cell Biology; Stead Family Department of Pediatrics; Epidemiology; Pediatric Dentistry; Craniofacial Anomalies Research Center; Dental Research
Record Identifier: 9984025453102771

Metrics

16 Record Views

24 Times Cited - Web of Science