Xiao Yu1, Natasha Nassar2, Pierpaolo Mastroiacovo3, Mark Canfield4, Boris Groisman5, Eva Bermejo-Sánchez6, Annukka Ritvanen7, Sonja Kiuru-Kuhlefelt7, Adriana Benavides8, Antonin Sipek9, Anna Pierini10, Fabrizio Bianchi10, Karin Källén11, Miriam Gatt12, Margery Morgan13, David Tucker13, M Aaurora Canessa14, Rosa Gajardo14, Osvaldo M Mutchinick15, Elena Szabova16, Melinda Csáky-Szunyogh17, Giovanna Tagliabue18, Janet D Cragan19, Wendy N Nembhard20, Anke Rissmann21, Dorit Goetz21, Carol Bower22, Gareth Baynam23, R Brian Lowry24, Juan A Leon25, Wei Luo25, Jocelyn Rouleau25, Ignacio Zarante26, Nicolas Fernandez27, Emmanuelle Amar28, Saeed Dastgiri29, Paolo Contiero30, Laura E Martínez-de-Villarreal31, Barry Borman32, J E H Bergman33, Hermien E K de Walle33, Charlotte A Hobbs34, Amy E Nance35, A J Agopian36. 1. School of Public Health, University of Texas Health Science Center at Houston, Houston, TX, USA. 2. Child Population and Translational Health Research, Children's Hospital at Westmead Clinical School, Faculty of Medicine and Health, University of Sydney, Sydney, Australia. 3. Centre of the International Clearinghouse for Birth Defects Surveillance and Research, Rome, Italy. 4. Birth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, TX, USA. 5. 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. 6. ECEMC, Centro de Investigación sobre Anomalías Congénitas (CIAC), Instituto de Investigación de Enfermedades Raras (IIER), Instituto de Salud Carlos III, Madrid, Spain. 7. Finnish Register of Congenital Malformations, National Institute for Health and Welfare, Helsinki, Finland. 8. Centro de Registro de Enfermedades Congénitas (CREC), Unidad de Enfermedades Congénitas, Instituto Costarricense de Investigación y Enseñanza en Nutrición y Salud (INCIENSA), Cartago, Costa Rica. 9. Department of Medical Genetics, Thomayer Hospital, Prague, Czech Republic. 10. Institute of Clinical Physiology, National Research Council (IFC-CNR), Pisa, Italy; Fondazione Toscana Gabriele Monasterio, Pisa, Italy. 11. National Board of Health and Social Welfare, Stockholm, Sweden. 12. Malta Congenital Anomalies Registry, Directorate for Health Information and Research, G'mangia Hill, G'mangia, Malta. 13. Congenital Anomaly Register & Information Service for Wales (CARIS), Public Health Wales, Swansea, Wales, UK. 14. Regional Register Congenital Malformation Maule Health Service (RRMC-SSM), Maule, Chile. 15. Department of Genetics, Registry and Epidemiological Surveillance of Congenital Malformations (RYVEMCE), Instituto Nacional de Ciencias Médicas y Nutrición "Salvador Zubirán", Mexico City, Mexico. 16. Slovak Medical University, FPH, Limbova, Bratislava, Slovak Republic. 17. Hungarian Congenital Abnormalities Registry and Rare Disease Centre, National Public Health Center, Budapest, Hungary. 18. Lombardy Cancer Registry Fondazione IRCCS Istituto Nazionale Tumori, Milan, Italy. 19. National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, GA, USA. 20. Fay W. Boozman College of Public Health, University of Arkansas for Medical Sciences and Arkansas Reproductive Health Monitoring System, Little Rock, AR, USA. 21. Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty Otto-von-Guericke University Magdeburg, Magdeburg, Germany. 22. Telethon Kids Institute, Subiaco, Western Australia, Australia. 23. Child Population and Translational Health Research, Children's Hospital at Westmead Clinical School, Faculty of Medicine and Health, University of Sydney, Sydney, Australia; Western Australian Register of Developmental Anomalies, Western Australian Department of Health, Perth, Australia. 24. Alberta Congenital Anomalies Surveillance System, Department Clinical Genetics, Alberta Children's Hospital, Calgary, AB, Canada. 25. Public Health Agency of Canada, Ottawa, Ontario, Canada. 26. Instituto de Genética Humana, Pontificia Universidad Javeriana, Bogotá, Colombia. 27. Division of Pediatric Urology, Hospital Universitario San Ignacio, Pontificia Universidad Javeriana, Bogotá, Colombia. 28. Registre Des Malformations En Rhone-Alpes, REMERA (ex Central-East France Register of Congenital Malformations, IEG), Lyon, France. 29. Tabriz Health Services Management Research Centre, Tabriz University of Medical Sciences, Tabriz, Iran. 30. Lombardy Birth Defect Registry, Fondazione IRCCS Istituto Nazionale dei Tumori, Milan, Italy. 31. Genetics Department, Medical School, Universidad Autónoma de Nuevo León, Nuevo León, Mexico. 32. New Zealand Birth Defects Registry, Massey University, Wellington, New Zealand. 33. Department of Genetics, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands. 34. Rady Children's Institute for Genomic Medicine, San Diego, CA, USA. 35. Utah Birth Defect Network, Division of Family Health and Preparedness, Utah Department of Health, Salt Lake City, UT, USA. 36. School of Public Health, University of Texas Health Science Center at Houston, Houston, TX, USA. Electronic address: a.j.agopian@uth.tmc.edu.
Abstract
BACKGROUND: Hypospadias is a common male birth defect that has shown widespread variation in reported prevalence estimates. Many countries have reported increasing trends over recent decades. OBJECTIVE: To analyze the prevalence and trends of hypospadias for 27 international programs over a 31-yr period. DESIGN, SETTING, AND PARTICIPANTS: The study population included live births, stillbirths, and elective terminations of pregnancy diagnosed with hypospadias during 1980-2010 from 27 surveillance programs around the world. OUTCOME MEASUREMENTS AND STATISTICAL ANALYSIS: We used joinpoint regression to analyze changes over time in international total prevalence of hypospadias across programs, prevalence for each specific program, and prevalence across different degrees of severity of hypospadias. RESULTS AND LIMITATIONS: The international total prevalence of hypospadias for all years was 20.9 (95% confidence interval: 19.2-22.6) per 10000 births. The prevalence for each program ranged from 2.1 to 39.1 per 10000 births. The international total prevalence increased 1.6 times during the study period, by 0.25 cases per 10000 births per year (p<0.05). When analyzed separately, there were increasing trends for first-, second-, and third-degree hypospadias during the early 1990s to mid-2000s. The majority of programs (61.9%) had a significantly increasing trend during many of the years evaluated. Limitations include known differences in data collection methods across programs. CONCLUSIONS: Although there have been changes in clinical practice and registry ascertainment over time in some countries, the consistency in the observed increasing trends across many programs and by degrees of severity suggests that the total prevalence of hypospadias may be increasing in many countries. This observation is contrary to some previous reports that suggested that the total prevalence of hypospadias was no longer increasing in recent decades. PATIENT SUMMARY: We report on the prevalence and trends of hypospadias among 27 birth defect surveillance systems, which indicate that the prevalence of hypospadias continues to increase internationally.
BACKGROUND:Hypospadias is a common male birth defect that has shown widespread variation in reported prevalence estimates. Many countries have reported increasing trends over recent decades. OBJECTIVE: To analyze the prevalence and trends of hypospadias for 27 international programs over a 31-yr period. DESIGN, SETTING, AND PARTICIPANTS: The study population included live births, stillbirths, and elective terminations of pregnancy diagnosed with hypospadias during 1980-2010 from 27 surveillance programs around the world. OUTCOME MEASUREMENTS AND STATISTICAL ANALYSIS: We used joinpoint regression to analyze changes over time in international total prevalence of hypospadias across programs, prevalence for each specific program, and prevalence across different degrees of severity of hypospadias. RESULTS AND LIMITATIONS: The international total prevalence of hypospadias for all years was 20.9 (95% confidence interval: 19.2-22.6) per 10000 births. The prevalence for each program ranged from 2.1 to 39.1 per 10000 births. The international total prevalence increased 1.6 times during the study period, by 0.25 cases per 10000 births per year (p<0.05). When analyzed separately, there were increasing trends for first-, second-, and third-degree hypospadias during the early 1990s to mid-2000s. The majority of programs (61.9%) had a significantly increasing trend during many of the years evaluated. Limitations include known differences in data collection methods across programs. CONCLUSIONS: Although there have been changes in clinical practice and registry ascertainment over time in some countries, the consistency in the observed increasing trends across many programs and by degrees of severity suggests that the total prevalence of hypospadias may be increasing in many countries. This observation is contrary to some previous reports that suggested that the total prevalence of hypospadias was no longer increasing in recent decades. PATIENT SUMMARY: We report on the prevalence and trends of hypospadias among 27 birth defect surveillance systems, which indicate that the prevalence of hypospadias continues to increase internationally.
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