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Prevalence and mortality in children with congenital diaphragmatic hernia: a multicountry study

Maria Politis 1 Eva Bermejo-Sánchez 2 Mark Canfield Paolo Contiero 3 Janet Cragan 4 Saeed Dastgiri 5 Hermien E.K. de Walle 6 Marcia Feldkamp 7 Amy Nance Boris Groisman Miriam Gatt Adriana Benavides-Lara Paula Hurtado-Villa 8 Kärin Kallén Danielle Landau Nathalie Lelong 9 Jorge Lopez-Camelo Laura Martinez 10 Margery Morgan Osvaldo Mutchinick Anna Pierini Anke Rissmann 11 Antonin Šípek Elena Szabova 12 Wladimir Wertelecki Ignacio Zarante 13 Marian Bakker 14 Vijaya Kancherla 15 Pierpaolo Mastroiacovo Wendy Nembhard 1 
Abstract : Purpose: This study determined the prevalence, mortality, and time trends of children with congenital diaphragmatic hernia (CDH). Methods: Twenty-five hospital- and population-based surveillance programs in 19 International Clearinghouse for Birth Defects Surveillance and Research member countries provided birth defects mortality data between 1974 and 2015. CDH cases included live births, stillbirths, or elective termination of pregnancy for fetal anomalies. Prevalence, cumulative mortality rates, and 95% confidence intervals (CIs) were calculated using Poisson regression and a Kaplan-Meier product-limit method. Joinpoint regression analyses were conducted to assess time trends. Results: The prevalence of CDH was 2.6 per 10,000 total births (95% CI: 2.5-2.7), slightly increasing between 2001 and 2012 (average annual percent change = 0.5%; 95% CI:-0.6 to 1.6). The total percent mortality of CDH was 37.7%, with hospital-based registries having more deaths among live births than population-based registries (45.1% vs. 33.8%). Mortality rates decreased over time (average annual percent change = -2.4%; 95% CI: -3.8 to 1.1). Most deaths due to CDH occurred among 2- to 6-day-old infants for both registry types (36.3%, hospital-based; 12.1%, population-based). Conclusions: The mortality of CDH has decreased over time. Mortality remains high during the first week and varied by registry type.
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Submitted on : Friday, June 11, 2021 - 10:50:39 AM
Last modification on : Wednesday, September 28, 2022 - 5:55:38 AM

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Maria Politis, Eva Bermejo-Sánchez, Mark Canfield, Paolo Contiero, Janet Cragan, et al.. Prevalence and mortality in children with congenital diaphragmatic hernia: a multicountry study. Annals of Epidemiology, Elsevier Masson, 2021, 56, pp.61-69.e3. ⟨10.1016/j.annepidem.2020.11.007⟩. ⟨hal-03257883⟩



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