Temporal and geographical variations in survival of children born with congenital anomalies in Europe: A multi-registry cohort study

Michele Santoro*, Alessio Coi, Anna Pierini, Judith Rankin, Svetlana Glinianaia, Joachim Tan, Abigail Reid, Ester Garne, Maria Loane, Joanne Given, Amaia Aizpurua, Gianni Astolfi, Ingeborg Barisic, Clara Cavero-Carbonell, Hermien E. K. de Walle, Elly Den Hond, Laura Garcia-Villodre, Miriam Gatt, Mika Gissler, Sue JordanBabak Khoshnood, Sonja Kiuru-Kuhlefelt, Kari Klungsoyr, Nathalie Lelong, Renee Lutke, Olatz Mokoroa, Vera Nelen, Amanda J. Neville, Ljubica Odak, Anke Rissmann, Ieuan Scanlon, Stine Kjaer Urhoj, Diana Wellesley, Wladimir Wertelecki, Lyubov Yevtushok, Joan K. Morris

*Bijbehorende auteur voor dit werk

OnderzoeksoutputAcademicpeer review

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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.

Originele taal-2English
Aantal pagina's12
TijdschriftPaediatric and Perinatal Epidemiology
DOI's
StatusE-pub ahead of print - 8-jun-2022

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