Maria D Politis1, Eva Bermejo-Sánchez2, Mark A Canfield3, Paolo Contiero4, Janet D Cragan5, Saeed Dastgiri6, Hermien E K de Walle7, Marcia L Feldkamp8, Amy Nance9, Boris Groisman10, Miriam Gatt11, Adriana Benavides-Lara12, Paula Hurtado-Villa13, Kärin Kallén14, Danielle Landau15, Nathalie Lelong16, Jorge Lopez-Camelo17, Laura Martinez18, Margery Morgan19, Osvaldo M Mutchinick20, Anna Pierini21, Anke Rissmann22, Antonin Šípek23, Elena Szabova24, Wladimir Wertelecki25, Ignacio Zarante26, Marian K Bakker7, Vijaya Kancherla27, Pierpaolo Mastroiacovo28, Wendy N Nembhard29. 1. Arkansas Center for Birth Defects Research and Prevention, and Department of Epidemiology, Fay W. Boozman College of Public Health, University of Arkansas for Medical Sciences, Little Rock, AR. 2. ECEMC (Spanish Collaborative Study of Congenital Malformations), CIAC (Research Center on Congenital Anomalies), Institute of Rare Diseases Research (IIER), Instituto de Salud Carlos III, Madrid, Spain. 3. Texas Department of State Health Services, Birth Defects Epidemiology and Surveillance Branch, Austin, TX. 4. Lombardy Congenital Anomalies Registry, Cancer Registry Unit, Fondazione IRCCS, Istituto Nazionale Tumori, Italy. 5. Metropolitan Atlanta Congenital Defects Program, National Center on Birth Defects and Development Disabilities, Centers for Disease Control and Prevention, Atlanta, GA. 6. Health Services Management Research Centre, Tabriz University of Medical Sciences, Tabriz, Iran. 7. Department of Genetics, University of Groningen, University Medical Center Groningen, Eurocat Northern Netherlands, Groningen, the Netherlands. 8. Division of Medical Genetics, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT. 9. Division of Family Health and Preparedness, Utah Department of Health, Utah Birth Defect Network, Bureau of Children with Special Health Care Needs, Salt Lake City, UT. 10. National Network of Congenital Anomalies of Argentina (RENAC), National Center of Medical Genetics, National Ministry of Health, Buenos Aires, Argentina. 11. Malta Congenital Anomalies Registry, Directorate for Health Information and Research, Malta. 12. Costa Rican Birth Defects Registry (CREC), Costa Rican Institute of Research and Education in Nutrition and Health (INCIENSA), Cartago, Costa Rica. 13. Department of Basic Sciences of Health, School of Health, Pontificia Universidad Javeriana Cali, Colombia. 14. National Board of Health and Welfare, Stockholm, Sweden. 15. Department of Neonatology, Soroka Medical Center, Beer-Sheva, Israel. 16. REMAPAR, Paris Registry of Congenital Malformations, Inserm UMR 1153, Obstetrical, Perinatal and Pediatric Epidemiology Research Team (Epopé), Center for Epidemiology and Statistics Sorbonne Paris Cité, DHU Risks in Pregnancy, Paris Descartes University, France. 17. ECLAMC, Center for Medical Education and Clinical Research (CEMIC-CONICET), Buenos Aires, Argentina. 18. Genetics Department, Hospital Universitario Dr Jose E. Gonzalez, Universidad Autonóma de Nuevo León, Mexico. 19. CARIS, the Congenital Anomaly Register for Wales, Singleton Hospital, Swansea, Wales, UK. 20. Department of Genetics, RYVEMCE, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, México City, Mexico. 21. Institute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio, Tuscany Registry of Congenital Defects, Pisa, Italy. 22. Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty, Otto-von-Guericke University, Magdeburg, Germany. 23. Department of Medical Genetics, Thomayer Hospital, Prague, Czech Republic. 24. Slovak Teratologic Information Centre (FPH), Slovak Medical University, Bratislava, Slovak Republic. 25. Omni-Net for Children International Charitable Fund Rivne, Ukraine. 26. Pontificia Universidad Javeriana, Bogotá, Colombia. 27. Department of Epidemiology, Emory University Rollins School of Public Health, Atlanta, GA. 28. International Center on Birth Defects, International Clearinghouse for Birth Defects Surveillance and Research, Rome, Italy. 29. Arkansas Center for Birth Defects Research and Prevention and Arkansas Reproductive Health Monitoring System and Department of Epidemiology, Fay W. Boozman College of Public Health, University of Arkansas for Medical Sciences, Little Rock, AR. Electronic address: wnnembhard@uams.edu.
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.
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.
Authors: V Kandice Mah; Mohammed Zamakhshary; Doug Y Mah; Brian Cameron; Juan Bass; Desmond Bohn; Leslie Scott; Sharifa Himidan; Mark Walker; Peter C W Kim Journal: J Pediatr Surg Date: 2009-05 Impact factor: 2.545
Authors: Juan Antonio Gili; Jorge Santiago López-Camelo; Wendy N Nembhard; Marian Bakker; Hermien E K de Walle; Erin B Stallings; Vijaya Kancherla; Paolo Contiero; Saeed Dastgiri; Marcia L Feldkamp; Amy Nance; Miriam Gatt; Laura Martínez; María Aurora Canessa; Boris Groisman; Paula Hurtado-Villa; Karin Källén; Danielle Landau; Nathalie Lelong; Margery Morgan; Jazmín Arteaga-Vázquez; Anna Pierini; Anke Rissmann; Antonin Sipek; Elena Szabova; Wladimir Wertelecki; Ignacio Zarante; Mark A Canfield; Pierpaolo Mastroiacovo Journal: Birth Defects Res Date: 2022-05-28 Impact factor: 2.661
Authors: Débora Gusmão Melo; Maria Teresa Vieira Sanseverino; Thanyse de Oliveira Schmalfuss; Mariela Larrandaburu Journal: Front Public Health Date: 2021-11-02