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

Vijaya Kancherla; Manasvi Sundar; Tandaki Lucita; Anke Lux; Marian K. Bakker; Jorieke E.H. Bergman; Eva Bermejo-Sánchez; Mark A. Canfield; Saeed Dastgiri; Marcia L. Feldkamp; Miriam Gatt; Boris Groisman; Paula Hurtado-Villa; Kärin Kallen; Danielle Landau; Nathalie Lelong; Jorge Lopez-Camelo; Laura Elia Martinez; Pierpaolo Mastroiacovo; Margery Morgan; Osvaldo M. Mutchinick; Amy E. Nance; Wendy N. Nembhard; Anna Pierini; Antonin Sipek; Erin B. Stallings; Elena Szabova; Giovanna Tagliabue; Wladimir Wertelecki; Ignacio Zarante; Anke Rissmann

doi:10.1002/bdr2.2129

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

Vijaya Kancherla, Manasvi Sundar, Tandaki Lucita, Anke Lux, Marian K. Bakker, Jorieke E.H. Bergman, Eva Bermejo-Sánchez, Mark A. Canfield, Saeed Dastgiri, Marcia L. Feldkamp, Miriam Gatt, Boris Groisman, Paula Hurtado-Villa, Kärin Kallen, Danielle Landau, Nathalie Lelong, Jorge Lopez-Camelo, Laura Elia Martinez, Pierpaolo Mastroiacovo, Margery MorganOsvaldo M. Mutchinick, Amy E. Nance, Wendy N. Nembhard, Anna Pierini, Antonin Sipek, Erin B. Stallings, Elena Szabova, Giovanna Tagliabue, Wladimir Wertelecki, Ignacio Zarante, Anke Rissmann^*

^*Corresponding author for this work

Reproductive Origins of Adult Health and Disease (ROAHD)

Research output: Contribution to journal › Article › Academic › peer-review

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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.

Original language	English
Pages (from-to)	390-404
Number of pages	15
Journal	Birth Defects Research
Volume	115
Issue number	3
Early online date	19-Nov-2022
DOIs	https://doi.org/10.1002/bdr2.2129
Publication status	Published - 1-Feb-2023

Keywords

anorectal atresia
birth defect
epidemiology
mortality
prevalence

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Prevalence and mortality among children with anorectal malformationFinal publisher's version, 2.31 MBLicence: CC BY-NC

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Kancherla, V., Sundar, M., Lucita, T., Lux, A., Bakker, M. K., Bergman, J. E. H., Bermejo-Sánchez, E., Canfield, M. A., Dastgiri, S., Feldkamp, M. L., Gatt, M., Groisman, B., Hurtado-Villa, P., Kallen, K., Landau, D., Lelong, N., Lopez-Camelo, J., Martinez, L. E., Mastroiacovo, P., ... Rissmann, A. (2023). Prevalence and mortality among children with anorectal malformation: A multi-country analysis. Birth Defects Research, 115(3), 390-404. https://doi.org/10.1002/bdr2.2129

@article{3f4b2fbce541424cb2e1b1d87cc1e8f8,

title = "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.",

keywords = "anorectal atresia, birth defect, epidemiology, mortality, prevalence",

author = "Vijaya Kancherla and Manasvi Sundar and Tandaki Lucita and Anke Lux and Bakker, {Marian K.} and Bergman, {Jorieke E.H.} and Eva Bermejo-S{\'a}nchez and Canfield, {Mark A.} and Saeed Dastgiri and Feldkamp, {Marcia L.} and Miriam Gatt and Boris Groisman and Paula Hurtado-Villa and K{\"a}rin Kallen and Danielle Landau and Nathalie Lelong and Jorge Lopez-Camelo and Martinez, {Laura Elia} and Pierpaolo Mastroiacovo and Margery Morgan and Mutchinick, {Osvaldo M.} and Nance, {Amy E.} and Nembhard, {Wendy N.} and Anna Pierini and Antonin Sipek and Stallings, {Erin B.} and Elena Szabova and Giovanna Tagliabue and Wladimir Wertelecki and Ignacio Zarante and Anke Rissmann",

note = "Publisher Copyright: {\textcopyright} 2022 The Authors. Birth Defects Research published by Wiley Periodicals LLC.",

year = "2023",

month = feb,

day = "1",

doi = "10.1002/bdr2.2129",

language = "English",

volume = "115",

pages = "390--404",

journal = "Birth Defects Research",

issn = "2472-1727",

publisher = "Wiley",

number = "3",

}

Kancherla, V, Sundar, M, Lucita, T, Lux, A, Bakker, MK , Bergman, JEH, Bermejo-Sánchez, E, Canfield, MA, Dastgiri, S, Feldkamp, ML, Gatt, M, Groisman, B, Hurtado-Villa, P, Kallen, K, Landau, D, Lelong, N, Lopez-Camelo, J, Martinez, LE, Mastroiacovo, P, Morgan, M, Mutchinick, OM, Nance, AE, Nembhard, WN, Pierini, A, Sipek, A, Stallings, EB, Szabova, E, Tagliabue, G, Wertelecki, W, Zarante, I & Rissmann, A 2023, 'Prevalence and mortality among children with anorectal malformation: A multi-country analysis', Birth Defects Research, vol. 115, no. 3, pp. 390-404. https://doi.org/10.1002/bdr2.2129

TY - JOUR

T1 - Prevalence and mortality among children with anorectal malformation

T2 - A multi-country analysis

AU - Kancherla, Vijaya

AU - Sundar, Manasvi

AU - Lucita, Tandaki

AU - Lux, Anke

AU - Bakker, Marian K.

AU - Bergman, Jorieke E.H.

AU - Bermejo-Sánchez, Eva

AU - Canfield, Mark A.

AU - Dastgiri, Saeed

AU - Feldkamp, Marcia L.

AU - Gatt, Miriam

AU - Groisman, Boris

AU - Hurtado-Villa, Paula

AU - Kallen, Kärin

AU - Landau, Danielle

AU - Lelong, Nathalie

AU - Lopez-Camelo, Jorge

AU - Martinez, Laura Elia

AU - Mastroiacovo, Pierpaolo

AU - Morgan, Margery

AU - Mutchinick, Osvaldo M.

AU - Nance, Amy E.

AU - Nembhard, Wendy N.

AU - Pierini, Anna

AU - Sipek, Antonin

AU - Stallings, Erin B.

AU - Szabova, Elena

AU - Tagliabue, Giovanna

AU - Wertelecki, Wladimir

AU - Zarante, Ignacio

AU - Rissmann, Anke

PY - 2023/2/1

Y1 - 2023/2/1

N2 - 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.

AB - 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.

KW - anorectal atresia

KW - birth defect

KW - epidemiology

KW - mortality

KW - prevalence

U2 - 10.1002/bdr2.2129

DO - 10.1002/bdr2.2129

M3 - Article

C2 - 36401554

AN - SCOPUS:85142275133

SN - 2472-1727

VL - 115

SP - 390

EP - 404

JO - Birth Defects Research

JF - Birth Defects Research

IS - 3

ER -

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

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