A Multicountry Analysis of Prevalence and Mortality among Neonates and Children with Bladder Exstrophy-Reference-Cited by-同舟云学术

A Multicountry Analysis of Prevalence and Mortality among Neonates and Children with Bladder Exstrophy

Published:2022-05-29 Issue: Volume: Page:
ISSN:0735-1631
Container-title:American Journal of Perinatology
language:en
Short-container-title:Am J Perinatol

Author:

Kancherla Vijaya¹,Tandaki Lucita²^ORCID,Sundar Manasvi¹,Lux Anke³,Bakker Marian K⁴,Bergman Jorieke EH⁴,Bermejo-Sánchez Eva⁵,Canfield Mark A⁶,Feldkamp Marcia L⁷,Groisman Boris⁸,Hurtado-Villa Paula⁹,Källén Karin¹⁰,Landau Danielle¹¹,Lelong Nathalie¹²,Lopez-Camelo Jorge¹³^ORCID,Mastroiacovo Pierpaolo¹⁴,Morgan Margery¹⁵,Mutchinick Osvaldo M¹⁶,Nance Amy E¹⁷,Nembhard Wendy N¹⁸,Pierini Anna¹⁹,Šípek Antonin²⁰,Stallings Erin B²¹,Szabova Elena²²,Wertelecki Wladimir²³,Zarante Ignacio²⁴,Rissmann Anke²^ORCID

Affiliation:

1. Department of Epidemiology, Emory University Rollins School of Public Health, Atlanta, Georgia

2. Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty, Otto-von-Guericke-University, Magdeburg, Germany

3. Institute for Biometrics and Medical Informatics, Medical Faculty, Otto-von-Guericke-University, Magdeburg, Germany

4. Department of Genetics, University of Groningen, University Medical Center Groningen, Eurocat Northern The Netherlands, Groningen, The Netherlands

5. ECEMC (Spanish Collaborative Study of Congenital Malformations), UIAC (Unidad de Investigación sobre Anomalías Congénitas), Instituto de Investigación de Enfermedades Raras (IIER). Instituto de Salud Carlos III. Madrid, Spain

6. Birth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, Texas

7. Division of Medical Genetics, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, Utah

8. National Network of Congenital Anomalies of Argentina (RENAC), National Center of Medical Genetics, National Administration of Laboratories and Health Institutes (ANLIS), National Ministry of Health, Buenos Aires, Argentina

9. Department of Basic Sciences of Health, School of Health, Pontificia Universidad Javeriana Cali, Colombia and Clínica Imbanaco, Cali, Colombia

10. National Board of Health and Welfare, Stockholm, Sweden

11. Department of Neonatology, Soroka Medical Center, Beer-Sheva, Israel

12. Université de Paris, Inserm U1153, Obstetrical, Perinatal and Pediatric Epidemiology Research Team (Epopé), Center for Epidemiology and Statistics Sorbonne Paris Cité (CRESS) Paris, France

13. ECLAMC, Center for Medical Education and Clinical Research (CEMIC-CONICET), Buenos Aires, Argentina

14. International Center on Birth Defects, International Clearinghouse for Birth Defects Surveillance and Research, Rome, Italy

15. CARIS, the Congenital Anomaly Register for Wales, Singleton Hospital, Swansea, Wales, United Kingdom

16. RYVEMCE (Registry and Epidemiologic Surveillance of Congenital Malformations), Department of Genetics, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Mexico City, Mexico

17. Utah Department of Health, Bureau of Children with Special Health Care Needs, Utah Birth Defects Network, Salt Lake City, Utah

18. Arkansas Center for Birth Defects Research and Prevention and Arkansas Reproductive Health Monitoring System, University of Arkansas for Medical Sciences, Fay Boozman College of Public Health, Department of Epidemiology, Little Rock, Arkansas

19. Institute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio, Tuscany Registry of Congenital Defects, Pisa, Italy

20. Department of Medical Genetics, Thomayer Hospital, Prague, Czech Republic

21. Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Developmental Disabilities, US Centers for Disease Control and Prevention, Atlanta, Georgia

22. Slovak Teratologic Information Centre (FPH), Slovak Medical University, Bratislava, Slovak Republic

23. OMNI-Net Ukraine, Rivne, Ukraine

24. Human Genetics Institute, Pontificia Universidad Javeriana, Bogotá, Colombia and Hospital Universitario San Ignacio, Bogotá, Colombia

Abstract

Objective Bladder exstrophy (BE) is a rare but severe birth defect affecting the lower abdominal wall and genitourinary system. The objective of the study is to examine the total prevalence, trends in prevalence, and age-specific mortality among individuals with BE. Study Design We conducted a retrospective cohort study. Data were analyzed from 20 birth defects surveillance programs, members of the International Clearinghouse for Birth Defects Surveillance and Research in 16 countries. Live births, stillbirths, and elective terminations of pregnancy for fetal anomaly (ETOPFA) diagnosed with BE from 1974 to 2014. Pooled and program-specific prevalence of BE per 100,000 total births was calculated. The 95% confidence intervals (CI) for prevalence were estimated using Poisson approximation of binomial distribution. Time trends in prevalence of BE from 2000 to 2014 were examined using Poisson regression. Proportion of deaths among BE cases was calculated on the day of birth, day 2 to 6, day 7 to 27, day 28 to 364, 1 to 4 years, and ≥5 years. Mortality analysis was stratified by isolated, multiple, and syndromic case status. Results The pooled total prevalence of BE was 2.58 per 100,000 total births (95% CI = 2.40, 2.78) for study years 1974 to 2014. Prevalence varied over time with a decreasing trend from 2000 to 2014. The first-week mortality proportion was 3.5, 17.3, and 14.6% among isolated, multiple, and syndromic BE cases, respectively. The majority of first-week mortality occurred on the first day of life among isolated, multiple, and syndromic BE cases. The proportion of first-week deaths was higher among cases reported from programs in Latin America where ETOPFA services were not available. Conclusions Prevalence of BE varied by program and showed a decreasing trend from 2000 to –2014. Mortality is a concern among multiple and syndromic cases, and a high proportion of deaths among cases occurred during the first week of life. Key Points

Publisher

Georg Thieme Verlag KG

Subject

Obstetrics and Gynecology,Pediatrics, Perinatology and Child Health

Link

http://www.thieme-connect.de/products/ejournals/pdf/10.1055/s-0042-1748318.pdf

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