Effectiveness of primary repair for low anorectal malformations in Uganda

Abstract

Background—Anorectal malformations (ARMs) have an incidence of up to 1 in 4000 live births and can require immediate neonatal surgery due to associated intestinal blockage. Due to limited surgical access, Ugandan children present late and undergo three separate staged operations: (1) initial colostomy formation; (2) repair of the ARM (called anoplasty); and (3) colostomy closure. Three operations result in long treatment duration, potential complications with each procedure, delays in care, and stigmata associated with colostomies. By offering primary repair for ARMs in a resource-limited setting, we expect to: reduce healthcare expenditure by families, length of treatment, length of hospital stay, frequency of hospital visits, and social rejection. Materials and methods—A pragmatic clinical trial was performed examining the effectiveness of primary repair (prospective arm) and comparing it with the three-stage repairs (retrospective arm). Results—Of the 241 patients included for analysis—157 patients had a three-stage repair, whereas 84 patients had one- or two-stage repair. The median [IQR age at the last surgery (days) was 730.0 (365.0, 1460.0) vs 180.0 (90.0, 285.0)] in three-stage and one- or two-stage repairs, respectively. There was no difference in postoperative complications compared to patients who had three-stage repair. Patients who had a two-stage repair had less time with colostomy than those with three-stage repair. Non-inferiority analysis demonstrated that the primary repair approach was non-inferior to the three-stage approach. Conclusions—Primary repair for ARM is effective in low-income settings. It allows for less time with colostomy with no difference in post-operative complications. The decision on approach for treatment depends on the surgeon’s experience and clinical judgment. Keywords Primary repair; Anorectal malformations; Pragmatic clinical trial; Uganda