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Prevalence of Active Long-term Problems in Patients With Anorectal Malformations: A Systematic Review
Laurie Rigueros Springford1, Martin J Connor, Katie Jones
11 Department of Pediatric and Neonatal Surgery, St. George's University Hospitals, National Health Service Foundation Trust, London, United Kingdom 2 Population Health Research Institute, St. George's University of London, London, United Kingdom.
Insights
Long-term issues like fecal incontinence and constipation are common in patients with anorectal malformations. Further research is needed to improve long-term outcomes and healthcare transitions for these individuals.
Area of Science:
- Pediatric Surgery
- Gastroenterology
- Urology
Background:
- Anorectal malformations (ARMs) are congenital rectal anomalies with high survival but variable long-term outcomes.
- Active problems in patients over 10 years old with ARMs remain understudied.
Purpose of the Study:
- To systematically determine the prevalence of common long-term active problems in patients with a history of anorectal malformation repair.
Main Methods:
- Systematic review of MEDLINE, EMBASE, and Cochrane Library databases (1994-2015).
- Included studies with outcome data for patients aged ≥10 years with ARMs (excluding cloaca).
- Analyzed prevalence of fecal, urinary, and sexual dysfunction using logit-transformed data.
Main Results:
- Twelve studies involving 455 patients were analyzed.
- Prevalence ranges: fecal incontinence (16.7%-76.7%), chronic constipation (22.2%-86.7%), urinary incontinence (1.7%-30.5%), ejaculatory dysfunction (15.6%-41.2%), erectile dysfunction (5.6%-11.8%).
Conclusions:
- Patients with ARMs experience a high prevalence of long-term functional issues into adolescence and adulthood.
- Limitations include retrospective design, small sample sizes, and heterogeneous patient populations.
- Multicenter research is essential to identify predictors of outcome and optimize follow-up care.
Background:
Anorectal malformations are a spectrum of congenital anomalies of the rectum with high infantile survival rates and variable outcomes. Long-term (>10 years old) active problems associated with this condition have been poorly investigated.
Objective:
The purpose of this review was to systematically define the prevalence of the most common active long-term problems in patients with a history of anorectal malformation repair.
Data Sources:
MEDLINE, EMBASE, and the Cochrane Library were searched electronically using the OVID search platform.
Study Selection:
Original articles from August 1, 1994, to October 20, 2015, that included outcome data for patients aged ≥10 years with anorectal malformation. Cloaca was excluded from the study.
Interventions:
Prevalence estimates of anorectal malformations were obtained from published articles. CIs were ascertained in the logit scale after transforming prevalence into log odds and were then transformed into the original scale. The same method was used for subgroup analysis investigating high and low anorectal malformations.
Main Outcome Measures:
The overall prevalences of fecal, urinary, and sexual dysfunction were analyzed.
Results:
Twelve studies including 455 patients with a history of anorectal malformation repair were included for analysis. The range of reported prevalence of long-term active problems was as follows: fecal incontinence, 16.7% to 76.7%; chronic constipation, 22.2% to 86.7%; urinary incontinence, 1.7% to 30.5%; ejaculatory dysfunction, 15.6% to 41.2%; and erectile dysfunction, 5.6% to 11.8%.
Limitations:
The study was limited by its retrospective, small size; multiple complex associated anomalies often not reported; and heterogeneous composition of patients with limited stratification analysis.
Conclusions:
There is an overall high prevalence of active long-term issues in adolescents and young adults with anorectal malformations. Additional multicenter research is needed to define characteristics and predictors of long-term outcome, to implement effective follow-up, and to transition to adult health care.
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