TY - JOUR
T1 - Temporal and geographical variations in survival of children born with congenital anomalies in Europe
T2 - A multi-registry cohort study
AU - Santoro, Michele
AU - Coi, Alessio
AU - Pierini, Anna
AU - Rankin, Judith
AU - Glinianaia, Svetlana
AU - Tan, Joachim
AU - Reid, Abigail
AU - Garne, Ester
AU - Loane, Maria
AU - Given, Joanne
AU - Aizpurua, Amaia
AU - Astolfi, Gianni
AU - Barisic, Ingeborg
AU - Cavero-Carbonell, Clara
AU - de Walle, Hermien E. K.
AU - Den Hond, Elly
AU - Garcia-Villodre, Laura
AU - Gatt, Miriam
AU - Gissler, Mika
AU - Jordan, Sue
AU - Khoshnood, Babak
AU - Kiuru-Kuhlefelt, Sonja
AU - Klungsoyr, Kari
AU - Lelong, Nathalie
AU - Lutke, Renee
AU - Mokoroa, Olatz
AU - Nelen, Vera
AU - Neville, Amanda J.
AU - Odak, Ljubica
AU - Rissmann, Anke
AU - Scanlon, Ieuan
AU - Urhoj, Stine Kjaer
AU - Wellesley, Diana
AU - Wertelecki, Wladimir
AU - Yevtushok, Lyubov
AU - Morris, Joan K.
PY - 2022/11
Y1 - 2022/11
N2 - 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.
AB - 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.
KW - cohort study
KW - congenital anomalies
KW - registry
KW - survival
KW - SYSTEMATIC ANALYSIS
KW - NEONATAL-MORTALITY
KW - INFANT-MORTALITY
KW - NATIONAL CAUSES
KW - GLOBAL BURDEN
KW - DISEASE
KW - TRENDS
KW - DEATH
U2 - 10.1111/ppe.12884
DO - 10.1111/ppe.12884
M3 - Article
SN - 0269-5022
VL - 36
SP - 792
EP - 803
JO - Paediatric and Perinatal Epidemiology
JF - Paediatric and Perinatal Epidemiology
IS - 6
ER -