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.


Journal

Journal of pediatric surgery
ISSN: 1531-5037
Titre abrégé: J Pediatr Surg
Pays: United States
ID NLM: 0052631

Informations de publication

Date de publication:
Sep 2022
Historique:
received: 22 07 2021
revised: 31 01 2022
accepted: 08 02 2022
pubmed: 24 3 2022
medline: 17 8 2022
entrez: 23 3 2022
Statut: ppublish

Résumé

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. 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. 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. 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 III.

Sections du résumé

BACKGROUND 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 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 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 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 METHODS
Level III.

Identifiants

pubmed: 35317943
pii: S0022-3468(22)00180-4
doi: 10.1016/j.jpedsurg.2022.02.015
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

89-96

Informations de copyright

Copyright © 2022 The Authors. Published by Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Declarations of Competing Interest None.

Auteurs

Hendrik J J van der Steeg (HJJ)

Department of Surgery-Pediatric Surgery, Amalia Children's Hospital, Radboud University Medical Center, Geert Grooteplein-Zuid 10, P.O. Box 9101, Nijmegen 6500 HB, the Netherland. Electronic address: herjan.vandersteeg@radboudumc.nl.

Iris A L M van Rooij (IALM)

Department for Health Evidence, Radboud Institute for Health Sciences, Radboud UMC, Nijmegen, the Netherland.

Barbara D Iacobelli (BD)

Department of Medical and Surgical Neonatology, Newborn Surgery Unit, Bambino Gesù Children's Hospital-Research Institute, Rome, Italy.

Cornelius E J Sloots (CEJ)

Department of Pediatric Surgery, Erasmus Medical Center Sophia Children's Hospital, Rotterdam, the Netherland.

Anna Morandi (A)

Department of Pediatric Surgery, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

Paul M A Broens (PMA)

Department of Surgery, Division of Pediatric Surgery, University Medical Center Groningen, Groningen, the Netherland.

Igor Makedonsky (I)

Department of Pediatric Surgery, Children's Hospital Dnepropetrovsk, Dnepropetrovsk, Ukraine.

Francesco Fascetti Leon (FF)

Department of Pediatric Surgery, University of Padua, Padua, Italy.

Eberhard Schmiedeke (E)

Department of Pediatric Surgery and Urology, Center for Child and Youth Health, Klinikum Bremen-Mitte, Bremen, Germany.

Araceli García Vázquez (AG)

Department of Pediatric Surgery, University Hospital 12 de Octubre, Madrid, Spain.

Marc Miserez (M)

Department of Abdominal Surgery, UZ Leuven, KU Leuven, Belgium.

Gabriele Lisi (G)

Department of Pediatric Surgery, University "Gabriele d'Annunzio " of Chieti-Pescara - "Santo Spirito" Hospital, Pescara, Italy.

Paola Midrio (P)

Department of Pediatric Surgery, Ca' Foncello Hospital, Treviso, Italy.

Eva E Amerstorfer (EE)

Department of Pediatric and Adolescent Surgery, Medical University of Graz, Austria.

Maria Fanjul (M)

Department of Pediatric Surgery, Hospital Gregorio Marañón, Madrid, Spain.

Johanna Ludwiczek (J)

Department of Pediatric Surgery, Kepler Universitätsklinikum GmbH, Linz, Austria.

Pernilla Stenström (P)

Department of Pediatric Surgery, Skane University Hospital, Lund University, Lund, Sweden.

Alida F W van der Steeg (AFW)

Department of Pediatric Surgery, Emma Children's Hospital, Amsterdam University Medical Center, Amsterdam, the Netherland; Department of Pediatric Surgery, Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherland.

Ivo de Blaauw (I)

Department of Surgery-Pediatric Surgery, Amalia Children's Hospital, Radboud University Medical Center, Geert Grooteplein-Zuid 10, P.O. Box 9101, Nijmegen 6500 HB, the Netherland.

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Classifications MeSH