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.

Autor: Mc Goldrick N; School of Dentistry, University of Dundee, Dundee, Scotland, UK., Revie G; School of Dentistry, University of Dundee, Dundee, Scotland, UK., Groisman B; 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., Hurtado-Villa P; Facultad de Ciencias de la Salud, Pontificia Universidad Javeriana, Cali, Colombia., Sipek A; Department of Medical Genetics, Thomayer University Hospital, Prague, Czech Republic., Khoshnood B; Université Paris Cité, Inserm, INRAE, Centre for Research in Epidemiology and StatisticS (CRESS), Obstetrical Perinatal and Pediatric Epidemiology Research Team, EPOPé, Paris, France., Rissmann A; Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty Otto-von-Guericke-University Magdeburg, Magdeburg, Germany., Dastgiri S; Tabriz Health Services Management Research Center, Tabriz University of Medical Sciences, Tabriz, Iran., Landau D; Israel Birth Defects Surveillance Program, NICU Soroka University Medical Center, Beer Sheva, Israel., Tagliabue G; Cancer Registry Unit, Fondazione IRCCS Istituto Nazionale dei Tumori, Lombardy birth defects registry, Lombardy, Italy., Pierini A; Institute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio, Tuscany Registry of Congenital Defects, Pisa, Italy., Gatt M; Malta Congenital Anomalies Registry, Directorate for Health Information and Research, Tal-Pietà, Malta., Mutchinick OM; Registry and Epidemiological Surveillance of Congenital Malformations (RYVEMCE), National Institute of Medical Sciences and Nutrition, Mexico City, Mexico., Martínez L; Genetics Department University Hospital, Universidad Autonoma de Nuevo Leon, Nuevo Leon, Mexico., de Walle HEK; Department of Genetics, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands., Szabova E; Faculty of Public Health, Slovak Medical University, Bratislava, Slovakia., Lopez Camelo J; ECLAMC: The Latin-American collaborative study of congenital malformations, Centro de Educación Médica e Investigaciones Clínicas (CEMIC-CONICET), Buenos Aires, Argentina., Källén K; Institute of Clinical Sciences, Lund University and National Board of Health and Welfare, Lund, Sweden., Morgan M; Congenital anomaly register and information service, Public Health Wales, Cardiff, Wales, UK., Wertelecki W; OMNI-Net Ukraine Birth Defects Program, Rivne, Ukraine., Nance A; Utah Department of Health, Utah Birth Defect Network, Salt Lake City, Utah, USA., Stallings EB; 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., Nembhard WN; Department of Epidemiology and The Arkansas Reproductive Health Monitoring System, University of Arkansas for Medical Sciences, Little Rock, Arkansas, USA., Mossey P; School of Dentistry, University of Dundee, Dundee, Scotland, UK.
Jazyk: angličtina
Zdroj: Birth defects research [Birth Defects Res] 2023 Jun 01; Vol. 115 (10), pp. 980-997. Date of Electronic Publication: 2023 Apr 26.
DOI: 10.1002/bdr2.2176
Abstrakt: Background: Cleft 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.
Methods: Data 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.
Results: The 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%.
Conclusions: Total 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.
(© 2023 The Authors. Birth Defects Research published by Wiley Periodicals LLC.)
Databáze: MEDLINE
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