Cleft lip Sidedness and the Association with Additional Congenital Malformations

Author:

Fell Matthew12ORCID,Fitzsimons Kate J.3ORCID,Hamilton Mark J.4,Medina Jibby3,Butterworth Sophie3ORCID,Park Min Hae5,Van der Meulen Jan5,Lewis Sarah2,Chong David6,Russell Craig JH7

Affiliation:

1. Spires Cleft Centre, John Radcliffe Hospital, Oxford, UK

2. The Cleft Collective, Bristol Dental School, University of Bristol, Bristol, UK

3. Cleft Registry and Audit Network, Clinical Effectiveness Unit, The Royal College of Surgeons of England, London UK

4. West of Scotland Centre for Genomic Medicine, Queen Elizabeth University Hospital, Glasgow UK

5. Department of Health Services Research and Policy, London School of Hygiene and Tropical Medicine, London UK

6. The Royal Children's Hospital, Melbourne, Australia

7. Royal Hospital for Children, Queen Elizabeth University Hospital, Glasgow, UK

Abstract

Objective To investigate the association between the sidedness of orofacial clefts and additional congenital malformations. Design Linkage of a national registry of cleft births to national administrative data of hospital admissions Setting National Health Service, England Participants 2007 children born with cleft lip ± alveolus (CL ± A) and 2724 with cleft lip and palate (CLP) born between 2000 and 2012. Main outcome measure The proportion of children with ICD-10 codes for additional congenital malformations by the sidedness (left, right or bilateral) of orofacial clefts. Results For CL ± A phenotypes, there was no evidence for a difference in the prevalence of additional anomalies between left (22%, reference), right (22%, aOR 1.02, 95% CI 0.80 to 1.28; P = .90) and bilateral clefts (23%, aOR 1.09, 95% CI 0.75 to 1.57; P = .66). For CLP phenotypes, there was evidence of a lower prevalence of additional malformations in left (23%, reference) compared to right (32%, aOR 1.54, 95% CI 1.25 to 1.91; P < .001) and bilateral clefts (33%, aOR 1.64, 95% CI 1.35 to 1.99; P < .001). Conclusions The prevalence of additional congenital malformations was similar across sidedness subtypes with CL ± A phenotypes but was different for sidedness subtypes within CLP cases. These data support the hypothesis that CL ± A has a different underlying aetiology from CLP and that within the CLP phenotype, right sided CLP may lie closer in aetiology to bilateral CLP than it does to left sided CLP.

Funder

Medical Research Council

Publisher

SAGE Publications

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