{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T18:39:08Z","timestamp":1789756748006,"version":"4.0.1"},"reference-count":36,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2022,6,8]],"date-time":"2022-06-08T00:00:00Z","timestamp":1654646400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Paediatric Perinatal Epid"],"published-print":{"date-parts":[[2022,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Congenital anomalies are a major cause of perinatal, neonatal and infant mortality.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>The aim was to investigate temporal changes and geographical variation in survival of children with major congenital anomalies (CA) in different European areas.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>In this population\u2010based linkage cohort study, 17 CA registries members of EUROCAT, the European network for the surveillance of CAs, successfully linked data on 115,219 live births with CAs to mortality records. Registries estimated Kaplan\u2013Meier survival at 28\u2009days and 5\u2009years of age and fitted Cox's proportional hazards models comparing mortality at 1\u2009year and 1\u20139\u2009years of age for children born during 2005\u20132014 with those born during 1995\u20132004. The hazard ratios (HR) from each registry were combined centrally using a random\u2010effects model. The 5\u2010year survival conditional on having survived to 28\u2009days of age was calculated.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The overall risk of death by 1\u2009year of age for children born with any major CA in 2005\u20132014 decreased compared to 1995\u20132004 (HR 0.68, 95% confidence interval [CI] 0.53, 0.89). Survival at 5\u2009years of age ranged between registries from 97.6% to 87.0%. The lowest survival was observed for the registry of OMNI\u2010Net (Ukraine) (87.0%, 95% CI 86.1, 87.9).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Survival of children with CAs improved for births in 2005\u20132014 compared with 1995\u20132004. The use of CA registry data linked to mortality data enables investigation of survival of children with CAs. Factors such as defining major CAs, proportion of terminations of pregnancy for foetal anomaly, source of mortality data and linkage methods are important to consider in the design of future studies and in the interpretation of the results on survival of children with CAs.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/ppe.12884","type":"journal-article","created":{"date-parts":[[2022,6,8]],"date-time":"2022-06-08T11:39:56Z","timestamp":1654688396000},"page":"792-803","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Temporal and geographical variations in survival of children born with congenital anomalies in Europe: A multi\u2010registry cohort study"],"prefix":"10.1111","volume":"36","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0676-3036","authenticated-orcid":false,"given":"Michele","family":"Santoro","sequence":"first","affiliation":[{"name":"Unit of Epidemiology of Rare diseases and Congenital anomalies, Institute of Clinical Physiology National Research Council  Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessio","family":"Coi","sequence":"additional","affiliation":[{"name":"Unit of Epidemiology of Rare diseases and Congenital anomalies, Institute of Clinical Physiology National Research Council  Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Pierini","sequence":"additional","affiliation":[{"name":"Unit of Epidemiology of Rare diseases and Congenital anomalies, Institute of Clinical Physiology National Research Council  Pisa Italy"},{"name":"Fondazione Toscana Gabriele Monasterio  Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judith","family":"Rankin","sequence":"additional","affiliation":[{"name":"Population Health Sciences Institute, Faculty of Medical Sciences Newcastle University  Newcastle upon Tyne UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Svetlana V.","family":"Glinianaia","sequence":"additional","affiliation":[{"name":"Population Health Sciences Institute, Faculty of Medical Sciences Newcastle University  Newcastle upon Tyne UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joachim","family":"Tan","sequence":"additional","affiliation":[{"name":"Population Health Research Institute, St George's University of London  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abigail","family":"Reid","sequence":"additional","affiliation":[{"name":"Population Health Research Institute, St George's University of London  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ester","family":"Garne","sequence":"additional","affiliation":[{"name":"Paediatric Department Hospital Lillebaelt  Kolding Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Loane","sequence":"additional","affiliation":[{"name":"Faculty of Life and Health Sciences Ulster University  Coleraine UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4921-1944","authenticated-orcid":false,"given":"Joanne","family":"Given","sequence":"additional","affiliation":[{"name":"Faculty of Life and Health Sciences Ulster University  Coleraine UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amaia","family":"Aizpurua","sequence":"additional","affiliation":[{"name":"Public Health Division of Gipuzkoa BioDonostia Research Institute  San Sebastian Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gianni","family":"Astolfi","sequence":"additional","affiliation":[{"name":"IMER Registry Department of Neuroscience and Rehabilitation University of Ferrara  Ferrara Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ingeborg","family":"Barisic","sequence":"additional","affiliation":[{"name":"Children's Hospital Zagreb, Centre of Excellence for Reproductive and Regenerative Medicine Medical School University of Zagreb  Zagreb Croatia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clara","family":"Cavero\u2010Carbonell","sequence":"additional","affiliation":[{"name":"Rare Diseases Research Unit Foundation for the Promotion of Health and Biomedical Research in the Valencian Region  Valencia Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hermien E. 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