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

• Autores: Kancherla, Vijaya; Sundar, Manasvi; Tandaki, Lucita; Lux, Anke; Bakker, Marian K; Bergman, Jorieke EH; Bermejo-Sánchez, Eva; Canfield, Mark A.; Dastgiri, Saeed; Feldkamp, Marcia L.; Gatt, Miriam; Groisman, Boris; Hurtado Villa, Paula; Kallen, Kärin; Landau, Danielle; Lelong, Nathalie; López Camelo, Jorge Santiago; Martinez, Laura Elia; Mastroiacovo, Pierpaolo; Morgan, Margery; Mutchinick, Osvaldo M.; Nance, Amy E.; Nembhard, Wendy N.; Pierini, Anna; Sipek, Antonin; Stallings, Erin B.; Szabova, Elena; Tagliabue, Giovanna; Wertelecki, Wladimir; Zarante, Ignacio; Rissmann, Anke
• Año de publicación: 2023
• Idioma: inglés
• Tipo de recurso: artículo
• Materia: ANORECTAL ATRESIA; BIRTH DEFECT; EPIDEMIOLOGY; MORTALITY; PREVALENCE
• Institución: Consejo Nacional de Investigaciones Científicas y Técnicas

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.
Fil: Kancherla, Vijaya. University of Emory; Estados Unidos
Fil: Sundar, Manasvi. University of Emory; Estados Unidos
Fil: Tandaki, Lucita. Otto-von-Guericke-University; Alemania
Fil: Lux, Anke. Otto-von-Guericke-University; Alemania
Fil: Bakker, Marian K. University of Groningen; Países Bajos
Fil: Bergman, Jorieke EH. University of Groningen; Países Bajos
Fil: Bermejo-Sánchez, Eva. Instituto de Salud Carlos III; España
Fil: Canfield, Mark A.. Texas Department of State Health Services; Estados Unidos
Fil: Dastgiri, Saeed. Tabriz Health Services Management Research Center; Irán
Fil: Feldkamp, Marcia L.. University of Utah; Estados Unidos
Fil: Gatt, Miriam. Directorate For Health Information And Research; Malta
Fil: Groisman, Boris. Dirección Nacional de Instituto de Investigación. Administración Nacional de Laboratorios e Institutos de Salud "Dr. Carlos G. Malbrán". Centro Nacional de Genética Médica; Argentina. Consejo Nacional de Investigaciones Científicas y Técnicas; Argentina
Fil: Hurtado Villa, Paula. Pontificia Universidad Javeriana; Colombia
Fil: Kallen, Kärin. National Board of Health and Welfare; Suecia
Fil: Landau, Danielle. Soroka University Medical Center; Israel
Fil: Lelong, Nathalie. Center for Epidemiology and Statistics Sorbonne Paris Cité; Francia
Fil: López Camelo, Jorge Santiago. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Parque Centenario. CEMIC-CONICET. Centro de Educaciones Médicas e Investigaciones Clínicas "Norberto Quirno". CEMIC-CONICET; Argentina
Fil: Martinez, Laura Elia. Universidad Autónoma de Nuevo León; México
Fil: Mastroiacovo, Pierpaolo. International Clearinghouse for Birth Defects Surveillance and Research; Italia
Fil: Morgan, Margery. Singleton Hospital; Reino Unido
Fil: Mutchinick, Osvaldo M.. Instituto Nacional de la Nutrición Salvador Zubiran; México
Fil: Nance, Amy E.. University of Utah; Estados Unidos
Fil: Nembhard, Wendy N.. Arkansas Center For Birth Defects Research And Prevention; Estados Unidos
Fil: Pierini, Anna. Gabriele Monasterio Foundation; Italia
Fil: Sipek, Antonin. Fakultni Thomayerova Nemocnice; República Checa
Fil: Stallings, Erin B.. Centers for Disease Control and Prevention; Estados Unidos
Fil: Szabova, Elena. Slovak Medical University; Eslovaquia
Fil: Tagliabue, Giovanna. Fondazione Irccs Istituto Nazionale Dei Tumori; Italia
Fil: Wertelecki, Wladimir. OMNI-Net Ukraine Programs; Ucrania
Fil: Zarante, Ignacio. Pontificia Universidad Javeriana; Colombia
Fil: Rissmann, Anke. Otto-von-Guericke-University; Alemania