Temporal and geographical variations in survival of children born with congenital anomalies in Europe: A multi-registry cohort study.
cohort study
congenital anomalies
registry
survival
Journal
Paediatric and perinatal epidemiology
ISSN: 1365-3016
Titre abrégé: Paediatr Perinat Epidemiol
Pays: England
ID NLM: 8709766
Informations de publication
Date de publication:
11 2022
11 2022
Historique:
revised:
22
03
2022
received:
10
12
2021
accepted:
03
04
2022
pubmed:
9
6
2022
medline:
21
10
2022
entrez:
8
6
2022
Statut:
ppublish
Résumé
Congenital anomalies are a major cause of perinatal, neonatal and infant mortality. The aim was to investigate temporal changes and geographical variation in survival of children with major congenital anomalies (CA) in different European areas. In this population-based 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-Meier survival at 28 days and 5 years of age and fitted Cox's proportional hazards models comparing mortality at 1 year and 1-9 years of age for children born during 2005-2014 with those born during 1995-2004. The hazard ratios (HR) from each registry were combined centrally using a random-effects model. The 5-year survival conditional on having survived to 28 days of age was calculated. The overall risk of death by 1 year of age for children born with any major CA in 2005-2014 decreased compared to 1995-2004 (HR 0.68, 95% confidence interval [CI] 0.53, 0.89). Survival at 5 years of age ranged between registries from 97.6% to 87.0%. The lowest survival was observed for the registry of OMNI-Net (Ukraine) (87.0%, 95% CI 86.1, 87.9). Survival of children with CAs improved for births in 2005-2014 compared with 1995-2004. 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.
Sections du résumé
BACKGROUND
Congenital anomalies are a major cause of perinatal, neonatal and infant mortality.
OBJECTIVES
The aim was to investigate temporal changes and geographical variation in survival of children with major congenital anomalies (CA) in different European areas.
METHODS
In this population-based 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-Meier survival at 28 days and 5 years of age and fitted Cox's proportional hazards models comparing mortality at 1 year and 1-9 years of age for children born during 2005-2014 with those born during 1995-2004. The hazard ratios (HR) from each registry were combined centrally using a random-effects model. The 5-year survival conditional on having survived to 28 days of age was calculated.
RESULTS
The overall risk of death by 1 year of age for children born with any major CA in 2005-2014 decreased compared to 1995-2004 (HR 0.68, 95% confidence interval [CI] 0.53, 0.89). Survival at 5 years of age ranged between registries from 97.6% to 87.0%. The lowest survival was observed for the registry of OMNI-Net (Ukraine) (87.0%, 95% CI 86.1, 87.9).
CONCLUSIONS
Survival of children with CAs improved for births in 2005-2014 compared with 1995-2004. 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.
Identifiants
pubmed: 35675091
doi: 10.1111/ppe.12884
pmc: PMC9796712
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
792-803Subventions
Organisme : Medical Research Council
ID : MR/K02325X/1
Pays : United Kingdom
Informations de copyright
© 2022 The Authors. Paediatric and Perinatal Epidemiology published by John Wiley & Sons Ltd.
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