Long-term survival of children born with congenital anomalies: A systematic review and meta-analysis of population-based studies.

摘要: Background Following a reduction in global child mortality due to communicable diseases, the relative contribution of congenital anomalies is increasing. Although infant survival children born with has improved for many anomaly types recent decades, there less evidence on beyond infancy. We aimed systematically review, summarise, and quantify existing population-based data long-term individuals specific major examine factors associated survival. Methods findings Seven electronic databases (Medline, Embase, Scopus, PsycINFO, CINAHL, ProQuest Natural, Biological Science Collections), reference lists, citations included articles studies published 1 January 1995 30 April 2020 were searched. Screening eligibility, extraction, quality appraisal performed duplicate. original that reported (beyond year life) follow-up starting from birth English language as peer-reviewed papers. Studies heart defects (CHDs) excluded because systematic review CHD Meta-analysis was pool estimates, accounting trends over time. Of 10,888 identified articles, 55 (n = 367,801 live births) met inclusion criteria summarised narratively, 41 54,676) investigating eight (spina bifida [n 7,422], encephalocele 1,562], oesophageal atresia 6,303], biliary 3,877], diaphragmatic hernia 6,176], gastroschisis 4,845], Down syndrome by presence 22,317], trisomy 18 2,174]) meta-analysis. These covered years 1970 2015. Survival spina bifida, atresia, hernia, gastroschisis, an significantly time, pooled odds ratios (ORs) surviving per 10-year increase being OR 1.34 (95% confidence interval [95% CI] 1.24–1.46), 1.50 CI 1.38–1.62), 1.62 1.28–2.05), 1.57 1.37–1.81), 1.24 1.02–1.5), 1.99 1.67–2.37), respectively (p < 0.001 all, except [p 0.029]). There no observed improvement (OR 0.98, 95% 0.95–1.01, p 0.19) native liver 0.96, 0.88–1.03, 0.26). The additional structural anomalies, low weight, earlier most commonly predictors reduced any type. main limitation meta-analysis small number size cohorts, which limited predictive capabilities models resulting wide intervals. Conclusions This summarises estimates anomalies. report significant last few decades predict up 20 age those 2020. information important planning delivery specialised medical, social, education services counselling affected families. trial registered PROSPERO database (CRD42017074675).