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Bowel function and associated risk factors at preschool and early childhood age in children with anorectal malformation type rectovestibular fistula : An ARM-Net consortium study

van der Steeg, Hendrik J.J. ; van Rooij, Iris A.L.M. ; Iacobelli, Barbara D. ; Sloots, Cornelius E.J. ; Morandi, Anna ; Broens, Paul M.A. ; Makedonsky, Igor ; Leon, Francesco Fascetti ; Schmiedeke, Eberhard and Vázquez, Araceli García , et al. (2022) In Journal of Pediatric Surgery 57(9). p.89-96
Abstract

Background: Outcome of patients operated for anorectal malformation (ARM) type rectovestibular fistula (RVF) is generally considered to be good. However, large multi-center studies are scarce, mostly describing pooled outcome of different ARM-types, in adult patients. Therefore, counseling parents concerning the bowel function at early age is challenging. Aim of this study was to evaluate bowel function of RVF-patients at preschool/early childhood age and determine risk factors for poor functional outcome. Methods: A multi-center cohort study was performed. Patient characteristics, associated anomalies, sacral ratio, surgical procedures, post-reconstructive complications, one-year constipation, and Bowel Function Score (BFS) at 4–7... (More)

Background: Outcome of patients operated for anorectal malformation (ARM) type rectovestibular fistula (RVF) is generally considered to be good. However, large multi-center studies are scarce, mostly describing pooled outcome of different ARM-types, in adult patients. Therefore, counseling parents concerning the bowel function at early age is challenging. Aim of this study was to evaluate bowel function of RVF-patients at preschool/early childhood age and determine risk factors for poor functional outcome. Methods: A multi-center cohort study was performed. Patient characteristics, associated anomalies, sacral ratio, surgical procedures, post-reconstructive complications, one-year constipation, and Bowel Function Score (BFS) at 4–7 years of follow-up were registered. Groups with below normal (BFS < 17; subgroups ‘poor’ ≤ 11, and ‘fair’ 11 < BFS < 17) and good outcome (BFS ≥ 17) were formed. Univariable analyses were performed to detect risk factors for outcome. Results: The study included 111 RVF-patients. Median BFS was 16 (range 6–20). The ‘below normal’ group consisted of 61 patients (55.0%). Overall, we reported soiling, fecal accidents, and constipation in 64.9%, 35.1% and 70.3%, respectively. Bowel management was performed in 23.4% of patients. Risk factors for poor outcome were tethered cord and low sacral ratio, while sacral anomalies, low sacral ratio, prior enterostomy, post-reconstructive complications, and one-year constipation were for being on bowel management. Conclusions: Although median BFS at 4–7 year follow-up is nearly normal, the majority of patients suffers from some degree of soiling and constipation, and almost 25% needs bowel management. Several factors were associated with poor bowel function outcome and bowel management. Level of Evidence: Level III.

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Contribution to journal
publication status
published
subject
keywords
Anorectal malformation, ARM-Net, Bowel function score, Early childhood, Observational cohort-study, Preschool age, Rectovestibular fistula, Type of study
in
Journal of Pediatric Surgery
volume
57
issue
9
pages
89 - 96
publisher
Elsevier
external identifiers
  • pmid:35317943
  • scopus:85126618776
ISSN
0022-3468
DOI
10.1016/j.jpedsurg.2022.02.015
language
English
LU publication?
yes
id
4e08332a-ed0d-4671-9b12-139c549b9f65
date added to LUP
2022-06-08 12:09:54
date last changed
2024-06-13 13:41:42
@article{4e08332a-ed0d-4671-9b12-139c549b9f65,
  abstract     = {{<p>Background: Outcome of patients operated for anorectal malformation (ARM) type rectovestibular fistula (RVF) is generally considered to be good. However, large multi-center studies are scarce, mostly describing pooled outcome of different ARM-types, in adult patients. Therefore, counseling parents concerning the bowel function at early age is challenging. Aim of this study was to evaluate bowel function of RVF-patients at preschool/early childhood age and determine risk factors for poor functional outcome. Methods: A multi-center cohort study was performed. Patient characteristics, associated anomalies, sacral ratio, surgical procedures, post-reconstructive complications, one-year constipation, and Bowel Function Score (BFS) at 4–7 years of follow-up were registered. Groups with below normal (BFS &lt; 17; subgroups ‘poor’ ≤ 11, and ‘fair’ 11 &lt; BFS &lt; 17) and good outcome (BFS ≥ 17) were formed. Univariable analyses were performed to detect risk factors for outcome. Results: The study included 111 RVF-patients. Median BFS was 16 (range 6–20). The ‘below normal’ group consisted of 61 patients (55.0%). Overall, we reported soiling, fecal accidents, and constipation in 64.9%, 35.1% and 70.3%, respectively. Bowel management was performed in 23.4% of patients. Risk factors for poor outcome were tethered cord and low sacral ratio, while sacral anomalies, low sacral ratio, prior enterostomy, post-reconstructive complications, and one-year constipation were for being on bowel management. Conclusions: Although median BFS at 4–7 year follow-up is nearly normal, the majority of patients suffers from some degree of soiling and constipation, and almost 25% needs bowel management. Several factors were associated with poor bowel function outcome and bowel management. Level of Evidence: Level III.</p>}},
  author       = {{van der Steeg, Hendrik J.J. and van Rooij, Iris A.L.M. and Iacobelli, Barbara D. and Sloots, Cornelius E.J. and Morandi, Anna and Broens, Paul M.A. and Makedonsky, Igor and Leon, Francesco Fascetti and Schmiedeke, Eberhard and Vázquez, Araceli García and Miserez, Marc and Lisi, Gabriele and Midrio, Paola and Amerstorfer, Eva E. and Fanjul, Maria and Ludwiczek, Johanna and Stenström, Pernilla and van der Steeg, Alida F.W. and de Blaauw, Ivo}},
  issn         = {{0022-3468}},
  keywords     = {{Anorectal malformation; ARM-Net; Bowel function score; Early childhood; Observational cohort-study; Preschool age; Rectovestibular fistula; Type of study}},
  language     = {{eng}},
  number       = {{9}},
  pages        = {{89--96}},
  publisher    = {{Elsevier}},
  series       = {{Journal of Pediatric Surgery}},
  title        = {{Bowel function and associated risk factors at preschool and early childhood age in children with anorectal malformation type rectovestibular fistula : An ARM-Net consortium study}},
  url          = {{http://dx.doi.org/10.1016/j.jpedsurg.2022.02.015}},
  doi          = {{10.1016/j.jpedsurg.2022.02.015}},
  volume       = {{57}},
  year         = {{2022}},
}