Prevalence and mortality among children with anorectal malformation: A multi-country analysis.

Autor: Kancherla V; Department of Epidemiology, Emory University Rollins School of Public Health, Atlanta, Georgia, USA., Sundar M; Department of Epidemiology, Emory University Rollins School of Public Health, Atlanta, Georgia, USA., Tandaki L; Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty, Otto-von-Guericke-University, Magdeburg, Germany., Lux A; Institute for Biometrics and Medical Informatics, Medical Faculty, Otto-von-Guericke-University, Magdeburg, Germany., Bakker MK; Department of Genetics, University of Groningen, University Medical Center Groningen, Eurocat Northern Netherlands, Groningen, The Netherlands., Bergman JE; Department of Genetics, University of Groningen, University Medical Center Groningen, Eurocat Northern Netherlands, Groningen, The Netherlands., Bermejo-Sánchez E; 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., Canfield MA; Birth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, Texas, USA., Dastgiri S; Tabriz Health Services Management Research Center, School of Medicine, Tabriz University of Medical Sciences, Tabriz, Iran., Feldkamp ML; Division of Medical Genetics, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, Utah, USA., Gatt M; Malta Congenital Anomalies Registry, Directorate for Health Information and Research, Guardamangia, Malta., Groisman B; 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., Hurtado-Villa P; Department of Basic Sciences of Health, School of Health, Pontificia Universidad Javeriana Cali, Cali, Colombia., Kallen K; National Board of Health and Welfare, Stockholm, Sweden., Landau D; Department of Neonatology, Soroka Medical Center, Beer-Sheva, Israel., Lelong N; Université de Paris, Inserm U1153, Obstetrical, Perinatal and Pediatric Epidemiology Research Team (Epopé), Center for Epidemiology and Statistics Sorbonne Paris Cité (CRESS), Paris, France., Lopez-Camelo J; ECLAMC, Center for Medical Education and Clinical Research (CEMIC-CONICET), Buenos Aires, Argentina., Martinez LE; Registro DAN (Registro de Defectos al Nacimiento), Departamento de Genética, Hospital Universitario Dr. José E. González, Universidad Autónoma de Nuevo León, Monterrey, Mexico., Mastroiacovo P; International Center on Birth Defects, International Clearinghouse for Birth Defects Surveillance and Research, Rome, Italy., Morgan M; CARIS, the Congenital Anomaly Register for Wales, Public Health Wales, Singleton Hospital, Swansea, UK., Mutchinick OM; RYVEMCE, Department of Genetics, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Mexico City, Mexico., Nance AE; Utah Department of Health, Bureau of Children with Special Health Care Needs, Utah Birth Defects Network, Salt Lake City, Utah, USA., Nembhard WN; Arkansas Center for Birth Defects Research and Prevention and Arkansas Reproductive Health Monitoring System, Fay Boozman College of Public Health, Department of Epidemiology, University of Arkansas for Medical Sciences, Little Rock, Arkansas, USA., Pierini A; Institute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio, Tuscany Registry of Congenital Defects, Pisa, Italy., Sipek A; Department of Medical Genetics, Thomayer Hospital, Prague, Czech Republic., Stallings EB; Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Development Disabilities, US Centers for Disease Control, Atlanta, Georgia, USA., Szabova E; Slovak Teratologic Information Centre (FPH), Slovak Medical University, Bratislava, Slovak Republic., Tagliabue G; Cancer Registry Unit, Fondazione IRCCS Istituto Nazionale dei Tumori, Lombardy, Italy., Wertelecki W; OMNI-Net Ukraine Programs, Rivne, Ukraine., Zarante I; Human Genetics Institute, Pontificia Universidad Javeriana, Bogotá, Colombia and Hospital Universitario San Ignacio, Bogotá, Colombia., Rissmann A; Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty, Otto-von-Guericke-University, Magdeburg, Germany.
Jazyk: angličtina
Zdroj: Birth defects research [Birth Defects Res] 2023 Feb 01; Vol. 115 (3), pp. 390-404. Date of Electronic Publication: 2022 Nov 19.
DOI: 10.1002/bdr2.2129
Abstrakt: Purpose: We examined the total prevalence, trends in prevalence, and age-specific mortality among individuals with anorectal malformation (ARM) METHODS: We conducted a retrospective cohort study using data from 24 population- and hospital-based birth defects surveillance programs affiliated with the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) from 18 countries and for births from 1974 to 2014. We estimated pooled and program-specific total prevalence per 10,000 total births. Poisson regression was used to assess time trends in prevalence from 2001 to 2012 when most programs contributed data. We calculated selected age-specific proportions of deaths, stratified by case status RESULTS: The pooled total prevalence of ARM was 3.26 per 10,000 total births (95% Confidence Interval = 3.19, 3.32) for birth years 1974-2014. About 60% of cases were multiple or syndromic. Prevalence of multiple, syndromic, and stillborn cases decreased from 2001 to 2012. The first week mortality proportion was 12.5%, 3.2%, 28.3%, and 18.2% among all, isolated, multiple, and syndromic cases, respectively CONCLUSIONS: ARM is relatively rare, with multiple and syndromic cases showing decreasing prevalence during the study period. Mortality is a concern during the first week of life, and especially among multiple and syndromic cases. Our descriptive epidemiological findings increase our understanding of geographic variation in the prevalence of ARM and can be used to plan needed clinical services. Exploring factors influencing prevalence and mortality among individuals with ARM could inform future studies.
(© 2022 The Authors. Birth Defects Research published by Wiley Periodicals LLC.)
Databáze: MEDLINE
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