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Prevalence and mortality among children with anorectal malformation: A multi-country analysis
V. Kancherla, M. Sundar, L. Tandaki, A. Lux, MK. Bakker, JE. Bergman, E. Bermejo-Sánchez, MA. Canfield, S. Dastgiri, ML. Feldkamp, M. Gatt, B. Groisman, P. Hurtado-Villa, K. Kallen, D. Landau, N. Lelong, J. Lopez-Camelo, LE. Martinez, P....
Jazyk angličtina Země Spojené státy americké
Typ dokumentu časopisecké články, práce podpořená grantem
Grantová podpora
CC999999
Intramural CDC HHS - United States
PubMed
36401554
DOI
10.1002/bdr2.2129
Knihovny.cz E-zdroje
- MeSH
- anorektální malformace * epidemiologie MeSH
- dítě MeSH
- lidé MeSH
- narození mrtvého plodu epidemiologie MeSH
- porod MeSH
- prevalence MeSH
- retrospektivní studie MeSH
- těhotenství MeSH
- Check Tag
- dítě MeSH
- lidé MeSH
- těhotenství MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- práce podpořená grantem MeSH
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.
Cancer Registry Unit Fondazione IRCCS Istituto Nazionale dei Tumori Lombardy Italy
CARIS the Congenital Anomaly Register for Wales Public Health Wales Singleton Hospital Swansea UK
Department of Epidemiology Emory University Rollins School of Public Health Atlanta Georgia USA
Department of Medical Genetics Thomayer Hospital Prague Czech Republic
Department of Neonatology Soroka Medical Center Beer Sheva Israel
ECEMC Instituto de Salud Carlos 3 Madrid Spain
ECLAMC Center for Medical Education and Clinical Research Buenos Aires Argentina
National Board of Health and Welfare Stockholm Sweden
OMNI Net Ukraine Programs Rivne Ukraine
Slovak Teratologic Information Centre Slovak Medical University Bratislava Slovak Republic
Citace poskytuje Crossref.org
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- $a Kancherla, Vijaya $u Department of Epidemiology, Emory University Rollins School of Public Health, Atlanta, Georgia, USA $1 https://orcid.org/0000000228038030
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- $a 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.
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