{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T16:40:59Z","timestamp":1791996059723,"version":"4.4.0"},"reference-count":21,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2023,4,26]],"date-time":"2023-04-26T00:00:00Z","timestamp":1682467200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2023,6]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Data from 22 population\u2010 and hospital\u2010based 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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>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%.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec>","DOI":"10.1002\/bdr2.2176","type":"journal-article","created":{"date-parts":[[2023,4,26]],"date-time":"2023-04-26T10:10:17Z","timestamp":1682503817000},"page":"980-997","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":24,"title":["A multi\u2010program analysis of cleft lip with cleft palate prevalence and mortality using data from 22 International Clearinghouse for Birth Defects Surveillance and Research programs, 1974\u20132014"],"prefix":"10.1002","volume":"115","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7353-1396","authenticated-orcid":false,"given":"Niall","family":"Mc Goldrick","sequence":"first","affiliation":[{"name":"School of Dentistry University of Dundee Dundee Scotland UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gavin","family":"Revie","sequence":"additional","affiliation":[{"name":"School of Dentistry University of Dundee Dundee Scotland UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6263-2562","authenticated-orcid":false,"given":"Boris","family":"Groisman","sequence":"additional","affiliation":[{"name":"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"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paula","family":"Hurtado\u2010Villa","sequence":"additional","affiliation":[{"name":"Facultad de Ciencias de la Salud Pontificia Universidad Javeriana Cali Colombia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonin","family":"Sipek","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics Thomayer University Hospital Prague Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Babak","family":"Khoshnood","sequence":"additional","affiliation":[{"name":"Universit\u00e9 Paris Cit\u00e9, Inserm, INRAE, Centre for Research in Epidemiology and StatisticS (CRESS) Obstetrical Perinatal and Pediatric Epidemiology Research Team, EPOP\u00e9 Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9437-2790","authenticated-orcid":false,"given":"Anke","family":"Rissmann","sequence":"additional","affiliation":[{"name":"Malformation Monitoring Centre Saxony\u2010Anhalt Medical Faculty Otto\u2010von\u2010Guericke\u2010University Magdeburg Magdeburg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saeed","family":"Dastgiri","sequence":"additional","affiliation":[{"name":"Tabriz Health Services Management Research Center Tabriz University of Medical Sciences Tabriz Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Danielle","family":"Landau","sequence":"additional","affiliation":[{"name":"Israel Birth Defects Surveillance Program NICU Soroka University Medical Center Beer Sheva Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giovanna","family":"Tagliabue","sequence":"additional","affiliation":[{"name":"Cancer Registry Unit, Fondazione IRCCS Istituto Nazionale dei Tumori Lombardy birth defects registry Lombardy Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Pierini","sequence":"additional","affiliation":[{"name":"Institute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio Tuscany Registry of Congenital Defects Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miriam","family":"Gatt","sequence":"additional","affiliation":[{"name":"Malta Congenital Anomalies Registry Directorate for Health Information and Research Tal\u2010Piet\u00e0 Malta"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Osvaldo M.","family":"Mutchinick","sequence":"additional","affiliation":[{"name":"Registry and Epidemiological Surveillance of Congenital Malformations (RYVEMCE), National Institute of Medical Sciences and Nutrition Mexico City Mexico"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Mart\u00ednez","sequence":"additional","affiliation":[{"name":"Genetics Department University Hospital Universidad Autonoma de Nuevo Leon Nuevo Leon Mexico"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hermein E.K.","family":"de Walle","sequence":"additional","affiliation":[{"name":"Department of Genetics University Medical Center Groningen, University of Groningen Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elena","family":"Szabova","sequence":"additional","affiliation":[{"name":"Faculty of Public Health Slovak Medical University Bratislava Slovakia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jorge","family":"Lopez Camelo","sequence":"additional","affiliation":[{"name":"ECLAMC: The Latin\u2010American collaborative study of congenital malformations, Centro de Educaci\u00f3n M\u00e9dica e Investigaciones Cl\u00ednicas (CEMIC\u2010CONICET) Buenos Aires Argentina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"ECEMC Peripheral Group","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin","family":"K\u00e4ll\u00e9n","sequence":"additional","affiliation":[{"name":"Institute of Clinical Sciences Lund University and National Board of Health and Welfare Lund Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margery","family":"Morgan","sequence":"additional","affiliation":[{"name":"Congenital anomaly register and information service Public Health Wales Cardiff Wales UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wladimir","family":"Wertelecki","sequence":"additional","affiliation":[{"name":"OMNI\u2010Net Ukraine Birth Defects Program Rivne Ukraine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4326-890X","authenticated-orcid":false,"given":"Amy","family":"Nance","sequence":"additional","affiliation":[{"name":"Utah Department of Health Utah Birth Defect Network Salt Lake City Utah USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erin B.","family":"Stallings","sequence":"additional","affiliation":[{"name":"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"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8318-4049","authenticated-orcid":false,"given":"Wendy N.","family":"Nembhard","sequence":"additional","affiliation":[{"name":"Department of Epidemiology and The Arkansas Reproductive Health Monitoring System University of Arkansas for Medical Sciences Little Rock Arkansas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Mossey","sequence":"additional","affiliation":[{"name":"School of Dentistry University of Dundee Dundee Scotland UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,4,26]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-642-30770-6"},{"key":"e_1_2_8_3_1","unstructured":"Calzolari E. 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