A multi‐program analysis of cleft lip with cleft palate prevalence and mortality using data from 22 International Clearinghouse for Birth Defects Surveillance and Research programs, 1974

A multi‐program analysis of cleft lip with cleft palate prevalence and mortality using data from 22 International Clearinghouse for Birth Defects Surveillance and Research programs, 1974–2014

Published:2023-04-26 Issue:10 Volume:115 Page:980-997
ISSN:2472-1727
Container-title:Birth Defects Research
language:en
Short-container-title:Birth Defects Research

Author:

Mc Goldrick Niall¹^ORCID,Revie Gavin¹,Groisman Boris²^ORCID,Hurtado‐Villa Paula³,Sipek Antonin⁴,Khoshnood Babak⁵,Rissmann Anke⁶^ORCID,Dastgiri Saeed⁷,Landau Danielle⁸,Tagliabue Giovanna⁹,Pierini Anna¹⁰,Gatt Miriam¹¹,Mutchinick Osvaldo M.¹²,Martínez Laura¹³,de Walle Hermein E.K.¹⁴,Szabova Elena¹⁵,Lopez Camelo Jorge¹⁶,Källén Karin¹⁷,Morgan Margery¹⁸,Wertelecki Wladimir¹⁹,Nance Amy²⁰^ORCID,Stallings Erin B.²¹,Nembhard Wendy N.²²^ORCID,Mossey Peter¹,

Affiliation:

1. School of Dentistry University of Dundee Dundee Scotland UK

2. National Network of Congenital Anomalies of Argentina (RENAC), National Institute of Epidemiology (INE), ANLIS, National Ministry of Health National Center of Medical Genetics Ciudad de Buenos Aires Argentina

3. Facultad de Ciencias de la Salud Pontificia Universidad Javeriana Cali Colombia

4. Department of Medical Genetics Thomayer University Hospital Prague Czech Republic

5. Université Paris Cité, Inserm, INRAE, Centre for Research in Epidemiology and StatisticS (CRESS) Obstetrical Perinatal and Pediatric Epidemiology Research Team, EPOPé Paris France

6. Malformation Monitoring Centre Saxony‐Anhalt Medical Faculty Otto‐von‐Guericke‐University Magdeburg Magdeburg Germany

7. Tabriz Health Services Management Research Center Tabriz University of Medical Sciences Tabriz Iran

8. Israel Birth Defects Surveillance Program NICU Soroka University Medical Center Beer Sheva Israel

9. Cancer Registry Unit, Fondazione IRCCS Istituto Nazionale dei Tumori Lombardy birth defects registry Lombardy Italy

10. Institute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio Tuscany Registry of Congenital Defects Pisa Italy

11. Malta Congenital Anomalies Registry Directorate for Health Information and Research Tal‐Pietà Malta

12. Registry and Epidemiological Surveillance of Congenital Malformations (RYVEMCE), National Institute of Medical Sciences and Nutrition Mexico City Mexico

13. Genetics Department University Hospital Universidad Autonoma de Nuevo Leon Nuevo Leon Mexico

14. Department of Genetics University Medical Center Groningen, University of Groningen Groningen the Netherlands

15. Faculty of Public Health Slovak Medical University Bratislava Slovakia

16. ECLAMC: The Latin‐American collaborative study of congenital malformations, Centro de Educación Médica e Investigaciones Clínicas (CEMIC‐CONICET) Buenos Aires Argentina

17. Institute of Clinical Sciences Lund University and National Board of Health and Welfare Lund Sweden

18. Congenital anomaly register and information service Public Health Wales Cardiff Wales UK

19. OMNI‐Net Ukraine Birth Defects Program Rivne Ukraine

20. Utah Department of Health Utah Birth Defect Network Salt Lake City Utah USA

21. Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Development Disabilities US Centers for Disease Control and Prevention Atlanta Georgia USA

22. Department of Epidemiology and The Arkansas Reproductive Health Monitoring System University of Arkansas for Medical Sciences Little Rock Arkansas USA

Abstract

AbstractBackgroundCleft lip with cleft palate (CLP) is a congenital condition that affects both the oral cavity and the lips. This study estimated the prevalence and mortality of CLP using surveillance data collected from birth defect registries around the world.MethodsData from 22 population‐ and hospital‐based surveillance programs affiliated with the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) in 18 countries on live births (LB), stillbirths (SB), and elective terminations of pregnancy for fetal anomaly (ETOPFA) for CLP from 1974 to 2014 were analyzed. Prevalence and survival (survival for LB only) estimates were calculated for total and subclassifications of CLP and by pregnancy outcome.ResultsThe pooled prevalence of total CLP cases was 6.4 CLP per 10,000 births. The prevalence of CLP and all of the pregnancy outcomes varied across programs. Higher ETOPFA rates were recorded in most European programs compared to programs in other continents. In programs reporting low ETOPFA rates or where there was no ascertainment of ETOPFA, the rate of CLP among LB and SB was higher compared to those where ETOPFA rates were ascertained. Overall survival for total CLP was 91%. For isolated CLP, the survival was 97.7%. CLP associated with multiple congenital anomalies had an overall survival of 77.1%, and for CLP associated with genetic/chromosomal syndromes, overall survival was 40.9%.ConclusionsTotal CLP prevalence reported in this study is lower than estimates from prior studies, with variation by pregnancy outcomes between programs. Survival was lower when CLP was associated with other congenital anomalies or syndromes compared to isolated CLP.

Publisher

Wiley

Subject

Health, Toxicology and Mutagenesis,Developmental Biology,Toxicology,Embryology,Pediatrics, Perinatology and Child Health

Link

https://onlinelibrary.wiley.com/doi/pdf/10.1002/bdr2.2176

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