Systematic Review of Reported Outcomes for Antegrade Continence Enema in Patients With Anorectal Malformation and

Signe Olsbø1, Sara George Kiserud2, Anders Telle Hoel3

  • 1Department of Gastrointestinal and Pediatric Surgery, Oslo University Hospital, Oslo, Norway; Institute of Clinical Medicine, University of Oslo, Norway.

PubMed

Insights

This review found significant variation in how antegrade continence enema (ACE) outcomes are reported for anorectal malformations (ARM) and Hirschsprung disease (HD). Validated questionnaires and patient-reported outcome measures (PROMs) are rarely used in ACE studies.

Area of Science:

  • Pediatric Surgery
  • Gastroenterology
  • Clinical Research Methodology

Background:

  • Antegrade continence enema (ACE) is a surgical procedure used to manage fecal incontinence in patients with anorectal malformations (ARM) and Hirschsprung disease (HD).
  • Standardized reporting of outcomes and study characteristics is crucial for evaluating treatment efficacy and guiding clinical practice.

Purpose of the Study:

  • To systematically identify and analyze the reported parameters and study characteristics in the existing literature on ACE procedures for patients with ARM and HD.
  • To assess the heterogeneity in outcome reporting and the utilization of patient-centered outcome measures in ACE studies.

Main Methods:

  • A comprehensive literature search was conducted following PRISMA guidelines across multiple databases (Ovid MEDLINE, Cochrane Central, Embase, CINAHL).
  • Search terms included "anorectal malformation" or "Hirschsprung disease" combined with relevant ACE procedure terms.
  • Data extraction and screening were performed independently by two reviewers, with specific inclusion and exclusion criteria applied.

Main Results:

  • Out of 257 identified papers, 48 met the inclusion criteria, predominantly retrospective studies (76%).
  • A high degree of heterogeneity was observed, with 301 different parameters identified, but only 9% reported in over 50% of publications.
  • Commonly reported baseline parameters included sex and age at surgery, while outcome parameters focused on enema success, complication rates, and revision surgery. Patient-reported outcome measures (PROMs) and quality of life assessments were infrequently utilized (25% and 6%, respectively).

Conclusions:

  • There is substantial heterogeneity in the reporting of outcomes for antegrade continence enema (ACE) in patients with ARM and HD.
  • The current literature rarely employs validated questionnaires or patient-reported outcome measures (PROMs), limiting comprehensive outcome assessment.
  • Standardization of reporting parameters and increased use of PROMs are recommended to improve the quality and comparability of research in this field.
Abstract