Analysis of Consanguinity as Risk Factor of Nonsyndromic Cleft Lips with or without Palate

Author:

Rochmah Yayun Siti1,Harumsari Stefani2,Christiono Sandy3,Hutami Islamy Rahma4,Fatimah-Muis Siti5,Faradz Sultana M.H.6

Affiliation:

1. Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Universitas Islam Sultan Agung, Semarang, Indonesia

2. Department of Medical Biology, Faculty of Medicine, Universitas Islam Sultan Agung, Semarang, Indonesia

3. Department of Pediatric Dentistry, Faculty of Dentistry, Universitas Islam Sultan Agung, Semarang, Indonesia

4. Department of Orthodontics, Faculty of Dentistry, Universitas Islam Sultan Agung, Semarang, Indonesia

5. Department of Nutrition, Faculty of Medicine, Universitas Diponegoro, Semarang, Indonesia

6. Center for Biomedical Research (CEBIOR), Faculty of Medicine, Universitas Diponegoro, Semarang, Indonesia

Abstract

Abstract Objectives The etiologies of nonsyndromic cleft lips with or without palate (NS CL/P) are multifactorial, which include consanguineous marriages. The incidence of NS CL/P is relatively high in Indonesia notably in one of Indonesia's tribes whose members frequently marry close cousins. Thus, the purpose of this study is to analysis consanguinity as risk factor of NS CL/P in Sasak tribe, East Lombok, Indonesia Materials and Methods An observational analysis was made of a collected database of NS CL/P patients treated in social services in regency hospital of Dr. Soejono Selong, East Lombok, Indonesia. Demographic data such as age, gender, address (urban/rural), parent's education, presence or absence of consanguinity, type of clefts, and a three-generation pedigree were collected by interview and hospital medical record. Before analysis, patient information was anonymized and deidentified. From 2016 to 2018, each of 100 cleft and normal subjects with their Sasak parent were audited. The risk factors were analyzed statistically using odds ratio (OR) and chi-squared test. Results Consanguineous marriages identified 54 cases (54%), and 10 cases (10%) out of a total each 100 NS CL/P and controls, respectively. The majority of consanguinity (53.7%) was discovered in marriages between first cousins. NS CL/P cases were statistically linked (p = 0.00) with consanguineous marriages (OR: 10; 95% confidence interval: 1.6–3.1); in which the most prevalent case is unilateral cleft lips. Conclusion Consanguineous marriage increases the risk of NS CL/P in Sasak tribe, East Lombok, Indonesia. The development of strategies to educate communities on the impacts of culture-consanguineous marriage is required. The genetic inheritance from their ancestor may be responsible for the increased incidence of NS CL/P.

Publisher

Georg Thieme Verlag KG

Subject

General Dentistry

Reference26 articles.

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5. The prevalence of non-syndromic orofacial clefts and associated congenital heart diseases of a tertiary hospital in Riyadh, Saudi Arabia;Z AlHammad;Saudi Dent J,2021

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