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...

Autores: Mc Goldrick, Niall; Revie, Gavin; Groisman, Boris; Hurtado Villa, Paula; Sipek, Antonin; Khoshnood, Babak; Rissmann, Anke; Dastgiri, Saeed; Landau, Danielle; Tagliabue, Giovanna; Pierini, Anna; Gatt, Miriam; Mutchinick, Osvaldo M.; Martínez, Laura; de Walle, Hermein E. K.; Szabova, Elena; López Camelo, Jorge Santiago; Källén, Karin; Morgan, Margery; Wertelecki, Wladimir; Nance, Amy; Stallings, Erin B.; Nembhard, Wendy N.; Mossey, Peter
Año de publicación: 2023
Idioma: inglés
Tipo de recurso: artículo
Estado: versión publicada
Descripción: 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.
Fil: Mc Goldrick, Niall. University of Dundee; Reino Unido
Fil: Revie, Gavin. University of Dundee; Reino Unido
Fil: Groisman, Boris. Dirección Nacional de Instituto de Investigación.Administración Nacional de Laboratorios e Institutos de Salud "Dr. Carlos G. Malbrán"; Argentina. Consejo Nacional de Investigaciones Científicas y Técnicas; Argentina
Fil: Hurtado Villa, Paula. Pontificia Universidad Javeriana; Colombia
Fil: Sipek, Antonin. Thomayer University Hospital; República Checa
Fil: Khoshnood, Babak. Universite de Paris. Umr - S1134 Biologie Integree Du Globule Rouge;
Fil: Rissmann, Anke. Otto-von-Guericke-Universität Magdeburg; Alemania
Fil: Dastgiri, Saeed. Tabriz University of Medical Sciences; Irán
Fil: Landau, Danielle. Soroka University Medical Center; Israel
Fil: Tagliabue, Giovanna. No especifíca;
Fil: Pierini, Anna. National Research Council and Fondazione Toscana Gabriele Monasterio; Italia
Fil: Gatt, Miriam. No especifíca;
Fil: Mutchinick, Osvaldo M.. Institute of Medical Sciences and Nutrition; México
Fil: Martínez, Laura. Universidad Autónoma de Nuevo León; México
Fil: de Walle, Hermein E. K.. University of Groningen; Países Bajos
Fil: Szabova, Elena. Slovak Medical University; Eslovaquia
Fil: López Camelo, Jorge Santiago. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Parque Centenario. CEMIC-CONICET. Centro de Educaciones Médicas e Investigaciones Clínicas "Norberto Quirno". CEMIC-CONICET; Argentina
Fil: Källén, Karin. Lund University and National Board of Health and Welfare; Suecia
Fil: Morgan, Margery. No especifíca;
Fil: Wertelecki, Wladimir. No especifíca;
Fil: Nance, Amy. No especifíca;
Fil: Stallings, Erin B.. No especifíca;
Fil: Nembhard, Wendy N.. No especifíca;
Fil: Mossey, Peter. University of Dundee; Reino Unido
Materia: craniofacial abnormalities
congenital anomalies,
mortality
prevalence
Nivel de accesibilidad: acceso abierto
Condiciones de uso: https://creativecommons.org/licenses/by/2.5/ar/
Repositorio
Institución: Consejo Nacional de Investigaciones Científicas y Técnicas
OAI Identificador: oai:ri.conicet.gov.ar:11336/256409

Acceder

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oai_identifier_str	oai:ri.conicet.gov.ar:11336/256409
network_acronym_str	CONICETDig
repository_id_str	3498
network_name_str	CONICET Digital (CONICET)
spelling	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, 1974–2014Mc Goldrick, NiallRevie, GavinGroisman, BorisHurtado Villa, PaulaSipek, AntoninKhoshnood, BabakRissmann, AnkeDastgiri, SaeedLandau, DanielleTagliabue, GiovannaPierini, AnnaGatt, MiriamMutchinick, Osvaldo M.Martínez, Laurade Walle, Hermein E. K.Szabova, ElenaLópez Camelo, Jorge SantiagoKällén, KarinMorgan, MargeryWertelecki, WladimirNance, AmyStallings, Erin B.Nembhard, Wendy N.Mossey, Petercraniofacial abnormalitiescongenital anomalies,mortalityprevalencehttps://purl.org/becyt/ford/3.3https://purl.org/becyt/ford/3Background: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.Fil: Mc Goldrick, Niall. University of Dundee; Reino UnidoFil: Revie, Gavin. University of Dundee; Reino UnidoFil: Groisman, Boris. Dirección Nacional de Instituto de Investigación.Administración Nacional de Laboratorios e Institutos de Salud "Dr. Carlos G. Malbrán"; Argentina. Consejo Nacional de Investigaciones Científicas y Técnicas; ArgentinaFil: Hurtado Villa, Paula. Pontificia Universidad Javeriana; ColombiaFil: Sipek, Antonin. Thomayer University Hospital; República ChecaFil: Khoshnood, Babak. Universite de Paris. Umr - S1134 Biologie Integree Du Globule Rouge;Fil: Rissmann, Anke. Otto-von-Guericke-Universität Magdeburg; AlemaniaFil: Dastgiri, Saeed. Tabriz University of Medical Sciences; IránFil: Landau, Danielle. Soroka University Medical Center; IsraelFil: Tagliabue, Giovanna. No especifíca;Fil: Pierini, Anna. National Research Council and Fondazione Toscana Gabriele Monasterio; ItaliaFil: Gatt, Miriam. No especifíca;Fil: Mutchinick, Osvaldo M.. Institute of Medical Sciences and Nutrition; MéxicoFil: Martínez, Laura. Universidad Autónoma de Nuevo León; MéxicoFil: de Walle, Hermein E. K.. University of Groningen; Países BajosFil: Szabova, Elena. Slovak Medical University; EslovaquiaFil: López Camelo, Jorge Santiago. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Parque Centenario. CEMIC-CONICET. Centro de Educaciones Médicas e Investigaciones Clínicas "Norberto Quirno". CEMIC-CONICET; ArgentinaFil: Källén, Karin. Lund University and National Board of Health and Welfare; SueciaFil: Morgan, Margery. No especifíca;Fil: Wertelecki, Wladimir. No especifíca;Fil: Nance, Amy. No especifíca;Fil: Stallings, Erin B.. No especifíca;Fil: Nembhard, Wendy N.. No especifíca;Fil: Mossey, Peter. University of Dundee; Reino UnidoWiley2023-04info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersionhttp://purl.org/coar/resource_type/c_6501info:ar-repo/semantics/articuloapplication/pdfapplication/pdfhttp://hdl.handle.net/11336/256409Mc Goldrick, Niall; Revie, Gavin; Groisman, Boris; Hurtado Villa, Paula; Sipek, Antonin; et al.; 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; Wiley; Birth Defects Research; 115; 10; 4-2023; 980-9972472-1727CONICET DigitalCONICETenginfo:eu-repo/semantics/altIdentifier/url/https://onlinelibrary.wiley.com/doi/10.1002/bdr2.2176info:eu-repo/semantics/altIdentifier/doi/10.1002/bdr2.2176info:eu-repo/semantics/openAccesshttps://creativecommons.org/licenses/by/2.5/ar/reponame:CONICET Digital (CONICET)instname:Consejo Nacional de Investigaciones Científicas y Técnicas2025-10-15T14:44:11Zoai:ri.conicet.gov.ar:11336/256409instacron:CONICETInstitucionalhttp://ri.conicet.gov.ar/Organismo científico-tecnológicoNo correspondehttp://ri.conicet.gov.ar/oai/requestdasensio@conicet.gov.ar; lcarlino@conicet.gov.arArgentinaNo correspondeNo correspondeNo correspondeopendoar:34982025-10-15 14:44:11.949CONICET Digital (CONICET) - Consejo Nacional de Investigaciones Científicas y Técnicasfalse
dc.title.none.fl_str_mv	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
title	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
spellingShingle	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 Mc Goldrick, Niall craniofacial abnormalities congenital anomalies, mortality prevalence
title_short	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
title_full	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
title_fullStr	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
title_full_unstemmed	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
title_sort	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
dc.creator.none.fl_str_mv	Mc Goldrick, Niall Revie, Gavin Groisman, Boris Hurtado Villa, Paula Sipek, Antonin Khoshnood, Babak Rissmann, Anke Dastgiri, Saeed Landau, Danielle Tagliabue, Giovanna Pierini, Anna Gatt, Miriam Mutchinick, Osvaldo M. Martínez, Laura de Walle, Hermein E. K. Szabova, Elena López Camelo, Jorge Santiago Källén, Karin Morgan, Margery Wertelecki, Wladimir Nance, Amy Stallings, Erin B. Nembhard, Wendy N. Mossey, Peter
author	Mc Goldrick, Niall
author_facet	Mc Goldrick, Niall Revie, Gavin Groisman, Boris Hurtado Villa, Paula Sipek, Antonin Khoshnood, Babak Rissmann, Anke Dastgiri, Saeed Landau, Danielle Tagliabue, Giovanna Pierini, Anna Gatt, Miriam Mutchinick, Osvaldo M. Martínez, Laura de Walle, Hermein E. K. Szabova, Elena López Camelo, Jorge Santiago Källén, Karin Morgan, Margery Wertelecki, Wladimir Nance, Amy Stallings, Erin B. Nembhard, Wendy N. Mossey, Peter
author_role	author
author2	Revie, Gavin Groisman, Boris Hurtado Villa, Paula Sipek, Antonin Khoshnood, Babak Rissmann, Anke Dastgiri, Saeed Landau, Danielle Tagliabue, Giovanna Pierini, Anna Gatt, Miriam Mutchinick, Osvaldo M. Martínez, Laura de Walle, Hermein E. K. Szabova, Elena López Camelo, Jorge Santiago Källén, Karin Morgan, Margery Wertelecki, Wladimir Nance, Amy Stallings, Erin B. Nembhard, Wendy N. Mossey, Peter
author2_role	author author author author author author author author author author author author author author author author author author author author author author author
dc.subject.none.fl_str_mv	craniofacial abnormalities congenital anomalies, mortality prevalence
topic	craniofacial abnormalities congenital anomalies, mortality prevalence
purl_subject.fl_str_mv	https://purl.org/becyt/ford/3.3 https://purl.org/becyt/ford/3
dc.description.none.fl_txt_mv	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. Fil: Mc Goldrick, Niall. University of Dundee; Reino Unido Fil: Revie, Gavin. University of Dundee; Reino Unido Fil: Groisman, Boris. Dirección Nacional de Instituto de Investigación.Administración Nacional de Laboratorios e Institutos de Salud "Dr. Carlos G. Malbrán"; Argentina. Consejo Nacional de Investigaciones Científicas y Técnicas; Argentina Fil: Hurtado Villa, Paula. Pontificia Universidad Javeriana; Colombia Fil: Sipek, Antonin. Thomayer University Hospital; República Checa Fil: Khoshnood, Babak. Universite de Paris. Umr - S1134 Biologie Integree Du Globule Rouge; Fil: Rissmann, Anke. Otto-von-Guericke-Universität Magdeburg; Alemania Fil: Dastgiri, Saeed. Tabriz University of Medical Sciences; Irán Fil: Landau, Danielle. Soroka University Medical Center; Israel Fil: Tagliabue, Giovanna. No especifíca; Fil: Pierini, Anna. National Research Council and Fondazione Toscana Gabriele Monasterio; Italia Fil: Gatt, Miriam. No especifíca; Fil: Mutchinick, Osvaldo M.. Institute of Medical Sciences and Nutrition; México Fil: Martínez, Laura. Universidad Autónoma de Nuevo León; México Fil: de Walle, Hermein E. K.. University of Groningen; Países Bajos Fil: Szabova, Elena. Slovak Medical University; Eslovaquia Fil: López Camelo, Jorge Santiago. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Parque Centenario. CEMIC-CONICET. Centro de Educaciones Médicas e Investigaciones Clínicas "Norberto Quirno". CEMIC-CONICET; Argentina Fil: Källén, Karin. Lund University and National Board of Health and Welfare; Suecia Fil: Morgan, Margery. No especifíca; Fil: Wertelecki, Wladimir. No especifíca; Fil: Nance, Amy. No especifíca; Fil: Stallings, Erin B.. No especifíca; Fil: Nembhard, Wendy N.. No especifíca; Fil: Mossey, Peter. University of Dundee; Reino Unido
description	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.
publishDate	2023
dc.date.none.fl_str_mv	2023-04
dc.type.none.fl_str_mv	info:eu-repo/semantics/article info:eu-repo/semantics/publishedVersion http://purl.org/coar/resource_type/c_6501 info:ar-repo/semantics/articulo
format	article
status_str	publishedVersion
dc.identifier.none.fl_str_mv	http://hdl.handle.net/11336/256409 Mc Goldrick, Niall; Revie, Gavin; Groisman, Boris; Hurtado Villa, Paula; Sipek, Antonin; et al.; 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; Wiley; Birth Defects Research; 115; 10; 4-2023; 980-997 2472-1727 CONICET Digital CONICET
url	http://hdl.handle.net/11336/256409
identifier_str_mv	Mc Goldrick, Niall; Revie, Gavin; Groisman, Boris; Hurtado Villa, Paula; Sipek, Antonin; et al.; 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; Wiley; Birth Defects Research; 115; 10; 4-2023; 980-997 2472-1727 CONICET Digital CONICET
dc.language.none.fl_str_mv	eng
language	eng
dc.relation.none.fl_str_mv	info:eu-repo/semantics/altIdentifier/url/https://onlinelibrary.wiley.com/doi/10.1002/bdr2.2176 info:eu-repo/semantics/altIdentifier/doi/10.1002/bdr2.2176
dc.rights.none.fl_str_mv	info:eu-repo/semantics/openAccess https://creativecommons.org/licenses/by/2.5/ar/
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rights_invalid_str_mv	https://creativecommons.org/licenses/by/2.5/ar/
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dc.publisher.none.fl_str_mv	Wiley
publisher.none.fl_str_mv	Wiley
dc.source.none.fl_str_mv	reponame:CONICET Digital (CONICET) instname:Consejo Nacional de Investigaciones Científicas y Técnicas
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repository.mail.fl_str_mv	dasensio@conicet.gov.ar; lcarlino@conicet.gov.ar
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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...

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