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

Hendrik J.J. van der Steeg*, Iris A.L.M. van Rooij, On-behalf-of-the-ARM-Net-Consortium, Barbara D. Iacobelli, Cornelius E.J. Sloots, Anna Morandi, Paul M.A. Broens, Igor Makedonsky, Francesco Fascetti Leon, Eberhard Schmiedeke, Araceli García Vázquez, Marc Miserez, Gabriele Lisi, Paola Midrio, Eva E. Amerstorfer, Maria Fanjul, Johanna Ludwiczek, Pernilla Stenström, Alida F.W. van der Steeg, Ivo de Blaauw

*Corresponding author for this work

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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 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.

Original languageEnglish
Pages (from-to)89-96
Number of pages8
JournalJournal of Pediatric Surgery
Volume57
Issue number9
DOIs
Publication statusPublished - Sep 2022

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