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Lookup NU author(s): Professor Judith Rankin, Dr Svetlana Glinianaia
This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License (CC BY-NC-ND).
© 2022 John Wiley & Sons Ltd. 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.
Author(s): Santoro M, Coi A, Pierini A, Rankin J, Glinianaia SV, Tan J, Reid A, Garne E, Loane M, Given J, Aizpurua A, Astolfi G, Barisic I, Cavero-Carbonell C, de Walle HEK, Den Hond E, Garcia-Villodre L, Gatt M, Gissler M, Jordan S, Khoshnood B, Kiuru-Kuhlefelt S, Klungsoyr K, Lelong N, Lutke R, Mokoroa O, Nelen V, Neville AJ, Odak L, Rissmann A, Scanlon I, Urhoj SK, Wellesley D, Wertelecki W, Yevtushok L, Morris JK
Publication type: Article
Publication status: Published
Journal: Paediatric and Perinatal Epidemiology
Year: 2022
Volume: 36
Issue: 6
Pages: 792-803
Print publication date: 01/11/2022
Online publication date: 08/06/2022
Acceptance date: 03/04/2022
Date deposited: 21/06/2022
ISSN (print): 0269-5022
ISSN (electronic): 1365-3016
Publisher: Wiley-Blackwell Publishing Ltd.
URL: https://doi.org/10.1111/ppe.12884
DOI: 10.1111/ppe.12884
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