Dataset Information

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

ABSTRACT:

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.

SUBMITTER: Kancherla V

PROVIDER: S-EPMC9898144 | biostudies-literature | 2023 Feb

REPOSITORIES: biostudies-literature

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

Kancherla Vijaya V Sundar Manasvi M Tandaki Lucita L Lux Anke A Bakker Marian K MK Bergman Jorieke Eh JE Bermejo-Sánchez Eva E Canfield Mark A MA Dastgiri Saeed S Feldkamp Marcia L ML Gatt Miriam M Groisman Boris B Hurtado-Villa Paula P Kallen Kärin K Landau Danielle D Lelong Nathalie N Lopez-Camelo Jorge J Martinez Laura Elia LE Mastroiacovo Pierpaolo P Morgan Margery M Mutchinick Osvaldo M OM Nance Amy E AE Nembhard Wendy N WN Pierini Anna A Sipek Antonin A Stallings Erin B EB Szabova Elena E Tagliabue Giovanna G Wertelecki Wladimir W Zarante Ignacio I Rissmann Anke A

Birth defects research 20221119 3

<h4>Purpose</h4>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 p ...[more]

PMID: 36401554

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Project description:Despite surgical correction, children with anorectal malformations may experience long-term bowel dysfunction, including fecal incontinence and/or disorders of evacuation. Anorectal manometry is the most widely used test of anorectal function. Although considerable attention has been devoted to its application in the anorectal malformation cohort, there have been few attempts to consolidate the findings obtained. This systematic review aimed to (1) synthesize and evaluate the existing data regarding anorectal manometry results in children following anorectal malformation repair, and (2) evaluate the manometry protocols utilized, including equipment, assessment approach, and interpretation. We reviewed four databases (Embase, MEDLINE, the Cochrane Library, and PubMed) for relevant articles published between 1 January 1985 and 10 March 2022. Studies reporting post-operative anorectal manometry in children (<18 years) following anorectal malformation repair were evaluated for eligibility. Sixty-three studies were eligible for inclusion. Of the combined total cohort of 2155 patients, anorectal manometry results were reported for 1755 children following repair of anorectal malformations. Reduced resting pressure was consistently identified in children with anorectal malformations, particularly in those with more complex malformation types and/or fecal incontinence. Significant variability was identified in relation to manometry equipment, protocols, and interpretation. Few studies provided adequate cohort medical characteristics to facilitate interpretation of anorectal manometry findings within the context of the broader continence mechanism. This review highlights a widespread lack of standardization in the anorectal manometry procedure used to assess anorectal function in children following anorectal malformation repair. Consequently, interpretation and comparison of findings, both within and between institutions, is exceedingly challenging, if not impossible. Standardized manometry protocols, accompanied by a consistent approach to analysis, including definitions of normality and abnormality, are essential to enhance the comparability and clinical relevance of results.

Project description:BackgroundThe evidence and method are limited for the associations between mortality and temperature variability (TV) within or between days.ObjectivesWe developed a novel method to calculate TV and investigated TV-mortality associations using a large multicountry data set.MethodsWe collected daily data for temperature and mortality from 372 locations in 12 countries/regions (Australia, Brazil, Canada, China, Japan, Moldova, South Korea, Spain, Taiwan, Thailand, the United Kingdom, and the United States). We calculated TV from the standard deviation of the minimum and maximum temperatures during the exposure days. Two-stage analyses were used to assess the relationship between TV and mortality. In the first stage, a Poisson regression model allowing over-dispersion was used to estimate the community-specific TV-mortality relationship, after controlling for potential confounders. In the second stage, a meta-analysis was used to pool the effect estimates within each country.ResultsThere was a significant association between TV and mortality in all countries, even after controlling for the effects of daily mean temperature. In stratified analyses, TV was still significantly associated with mortality in cold, hot, and moderate seasons. Mortality risks related to TV were higher in hot areas than in cold areas when using short TV exposures (0-1 days), whereas TV-related mortality risks were higher in moderate areas than in cold and hot areas when using longer TV exposures (0-7 days).ConclusionsThe results indicate that more attention should be paid to unstable weather conditions in order to protect health. These findings may have implications for developing public health policies to manage health risks of climate change.CitationGuo Y, Gasparrini A, Armstrong BG, Tawatsupa B, Tobias A, Lavigne E, Coelho MS, Pan X, Kim H, Hashizume M, Honda Y, Guo YL, Wu CF, Zanobetti A, Schwartz JD, Bell ML, Overcenco A, Punnasiri K, Li S, Tian L, Saldiva P, Williams G, Tong S. 2016. Temperature variability and mortality: a multi-country study. Environ Health Perspect 124:1554-1559; http://dx.doi.org/10.1289/EHP149.

Dataset Information

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

Purpose

Publications

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

Similar Datasets

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