Juan Antonio Gili1,2, Jorge Santiago López-Camelo1, Wendy N Nembhard3, Marian Bakker4, Hermien E K de Walle4, Erin B Stallings5, Vijaya Kancherla6, Paolo Contiero7, Saeed Dastgiri8, Marcia L Feldkamp9, Amy Nance10, Miriam Gatt11, Laura Martínez12, María Aurora Canessa13, Boris Groisman14, Paula Hurtado-Villa15, Karin Källén16, Danielle Landau17, Nathalie Lelong18, Margery Morgan19, Jazmín Arteaga-Vázquez20, Anna Pierini21, Anke Rissmann22, Antonin Sipek23, Elena Szabova24, Wladimir Wertelecki25, Ignacio Zarante26, Mark A Canfield27, Pierpaolo Mastroiacovo28. 1. ECLAMC, Centro de Educación Médica e Investigaciones Clínicas (CEMIC-CONICET), Buenos Aires, Argentina. 2. Instituto Académico Pedagógico de Ciencias Humanas, Universidad Nacional de Villa María, Córdoba, Argentina. 3. Department of Epidemiology, Arkansas Center for Birth Defects Research and Prevention and Arkansas Reproductive Health Monitoring System, University of Arkansas for Medical Sciences, Little Rock, Arkansas, USA. 4. Department of Genetics, University of Groningen, University Medical Center Groningen, EUROCAT Northern Netherlands, Groningen, The Netherlands. 5. Metro Atlanta Congenital Defects Program (MACDP), Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Developmental Disabilities, CDC, Atlanta, Georgia, USA. 6. Department of Epidemiology, Emory University Rollins School of Public Health, Atlanta, Georgia, USA. 7. Lombardy Congenital Anomalies Registry, Cancer Registry Unit, Fondazione IRCCS, Istituto Nazionale Tumori, Milan, Italy. 8. Health Services Management Research Centre, Tabriz University of Medical Sciences, Tabriz, Iran. 9. Division of Medical Genetics, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, Utah, USA. 10. Utah Birth Defect Network, Bureau of Children with Special Health Care Needs, Division of Family Health and Preparedness, Utah Department of Health, Salt Lake City, Utah, USA. 11. Malta Congenital Anomalies Registry, Directorate for Health Information and Research, Tal-Pietà, Malta. 12. Genetics Department, Hospital Universitario Dr. José E. González, Universidad Autonóma de Nuevo León, San Nicolás de los Garza, Mexico. 13. Regional Register Congenital Malformation Maule Health Service (RRMC-SSM), Maule, Chile. 14. 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 and Social Development, Buenos Aires, Argentina. 15. Department of Basic Sciences of Health, School of Health, Pontificia Universidad Javeriana, Cali, Colombia. 16. National Board of Health and Welfare, Stockholm, Sweden. 17. Department of Neonatology, Soroka Medical Center, Beer-Sheva, Israel. 18. Université de Paris, CRESS Obstetrical, Perinatal and Pediatric Epidemiology Research Team (EPOPé), INSERM, INRA, Paris, France. 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. Medical Faculty, Malformation Monitoring Centre Saxony-Anhalt, Otto-von-Guericke University, Magdeburg, Germany. 23. Department of Medical Genetics, Thomayer University 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. Human Genetics Institute, Pontificia Universidad Javeriana, Bogotá, Colombia. 27. Birth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, Texas, USA. 28. International Center on Birth Defects, International Clearinghouse for Birth Defects Surveillance and Research, Rome, Italy.
Abstract
BACKGROUND: Congenital hydrocephalus (CH) comprises a heterogeneous group of birth anomalies with a wide-ranging prevalence across geographic regions and registry type. The aim of the present study was to analyze the early neonatal case fatality rate (CFR) and total birth prevalence of newborns diagnosed with CH. METHODS: Data were provided by 25 registries from four continents participating in the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) on births ascertained between 2000 and 2014. Two CH rates were calculated using a Poisson distribution: early neonatal CFR (death within 7 days) per 100 liveborn CH cases (CFR) and total birth prevalence rate (BPR) per 10,000 births (including live births and stillbirths) (BPR). Heterogeneity between registries was calculated using a meta-analysis approach with random effects. Temporal trends in CFR and BPR within registries were evaluated through Poisson regression modeling. RESULTS: A total of 13,112 CH cases among 19,293,280 total births were analyzed. The early neonatal CFR was 5.9 per 100 liveborn cases, 95% confidence interval (CI): 5.4-6.8. The CFR among syndromic cases was 2.7 times (95% CI: 2.2-3.3) higher than among non-syndromic cases (10.4% [95% CI: 9.3-11.7] and 4.4% [95% CI: 3.7-5.2], respectively). The total BPR was 6.8 per 10,000 births (95% CI: 6.7-6.9). Stratified by elective termination of pregnancy for fetal anomalies (ETOPFA), region and system, higher CFR were observed alongside higher BPR rates. The early neonatal CFR and total BPR did not show temporal variation, with the exception of a CFR decrease in one registry. CONCLUSIONS: Findings of early neonatal CFR and total BPR were highly heterogeneous among registries participating in ICBDSR. Most registries with higher CFR also had higher BPR. Differences were attributable to type of registry (hospital-based vs. population-based), ETOPFA (allowed yes or no) and geographical regions. These findings contribute to the understanding of regional differences of CH occurrence and early neonatal deaths.
BACKGROUND: Congenital hydrocephalus (CH) comprises a heterogeneous group of birth anomalies with a wide-ranging prevalence across geographic regions and registry type. The aim of the present study was to analyze the early neonatal case fatality rate (CFR) and total birth prevalence of newborns diagnosed with CH. METHODS: Data were provided by 25 registries from four continents participating in the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) on births ascertained between 2000 and 2014. Two CH rates were calculated using a Poisson distribution: early neonatal CFR (death within 7 days) per 100 liveborn CH cases (CFR) and total birth prevalence rate (BPR) per 10,000 births (including live births and stillbirths) (BPR). Heterogeneity between registries was calculated using a meta-analysis approach with random effects. Temporal trends in CFR and BPR within registries were evaluated through Poisson regression modeling. RESULTS: A total of 13,112 CH cases among 19,293,280 total births were analyzed. The early neonatal CFR was 5.9 per 100 liveborn cases, 95% confidence interval (CI): 5.4-6.8. The CFR among syndromic cases was 2.7 times (95% CI: 2.2-3.3) higher than among non-syndromic cases (10.4% [95% CI: 9.3-11.7] and 4.4% [95% CI: 3.7-5.2], respectively). The total BPR was 6.8 per 10,000 births (95% CI: 6.7-6.9). Stratified by elective termination of pregnancy for fetal anomalies (ETOPFA), region and system, higher CFR were observed alongside higher BPR rates. The early neonatal CFR and total BPR did not show temporal variation, with the exception of a CFR decrease in one registry. CONCLUSIONS: Findings of early neonatal CFR and total BPR were highly heterogeneous among registries participating in ICBDSR. Most registries with higher CFR also had higher BPR. Differences were attributable to type of registry (hospital-based vs. population-based), ETOPFA (allowed yes or no) and geographical regions. These findings contribute to the understanding of regional differences of CH occurrence and early neonatal deaths.
Authors: C Scala; A Familiari; A Pinas; A T Papageorghiou; A Bhide; B Thilaganathan; A Khalil Journal: Ultrasound Obstet Gynecol Date: 2017-02-28 Impact factor: 7.299
Authors: Wendy N Nembhard; Jorieke E H Bergman; Maria D Politis; Jazmín Arteaga-Vázquez; Eva Bermejo-Sánchez; Mark A Canfield; Janet D Cragan; Saeed Dastgiri; Hermien E K de Walle; Marcia L Feldkamp; Amy Nance; Miriam Gatt; Boris Groisman; Paula Hurtado-Villa; Kärin Kallén; Danielle Landau; Nathalie Lelong; Jorge Lopez-Camelo; Laura Martinez; Margery Morgan; Anna Pierini; Anke Rissmann; Antonin Šípek; Elena Szabova; Giovanna Tagliabue; Wladimir Wertelecki; Ignacio Zarante; Marian K Bakker; Vijaya Kancherla; Pierpaolo Mastroiacovo Journal: Birth Defects Res Date: 2020-10-17 Impact factor: 2.661
Authors: Ignacio Zarante; Paula Hurtado-Villa; Salimah R Walani; Vijaya Kancherla; Jorge López Camelo; Roberto Giugliani; Boris Groisman; Christopher P Howson; Pablo Durán Journal: Rev Panam Salud Publica Date: 2019-02-14
Authors: Marian K Bakker; Vijaya Kancherla; Mark A Canfield; Eva Bermejo-Sanchez; Janet D Cragan; Saeed Dastgiri; Hermien E K De Walle; Marcia L Feldkamp; Boris Groisman; Miriam Gatt; Paula Hurtado-Villa; Karin Kallen; Daniella Landau; Nathalie Lelong; Jorge S Lopez Camelo; Laura Martínez; Margery Morgan; Osvaldo M Mutchinick; Wendy N Nembhard; Anna Pierini; Anke Rissmann; Antonin Sipek; Elena Szabova; Giovanna Tagliabue; Wladimir Wertelecki; Ignacio Zarante; Pierpaolo Mastroiacovo Journal: Paediatr Perinat Epidemiol Date: 2019-10-21 Impact factor: 3.980