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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.
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.
Aim:
To identify reported parameters and study characteristics in the literature on antegrade continence enema (ACE) in patients with anorectal malformations (ARM) and Hirschsprung disease (HD).
Methods:
Following PRISMA guidelines, we performed a search using the terms "anorectal malformation" or "Hirschsprung disease", combined with "appendicostomy," "antegrade continence enema," "ACE", "Malone", or "cecostomy" in the databases Ovid MEDLINE, Cochrane Central, Embase, and CINAHL. Two reviewers independently screened and extracted data. Exclusion criteria included <10 patients with ARM and/or HD, age at ACE >18 years, non-English papers, opinion articles, conference abstracts, reviews, and meta-analyses.
Results:
257 papers were identified, and 48 met the inclusion criteria. 76 % were retrospective studies and 9 % multi-center studies. Most studies (84 %) mixed three or more diagnoses, with a median of 16 ARM and 3 HD patients per study. A total of 301 different parameters were identified. Nine (3 %) parameters were mentioned in more than 50 % of the publications. The most frequent baseline parameters were sex (98 %) and age at surgery (88 %). Common outcome parameters included success of enema (92 %), complication rate (80 %) and revision surgery (79 %). Patient reported outcome measures were utilized in 12 (25 %) papers, and 3 (6 %) assessed quality of life.
Conclusion:
There is substantial heterogeneity when reporting ACE outcomes. The use of validated questionnaires and PROMs is rare.
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