During the 4-year period of the study, 36 consecutive patients with rectoperineal fistula were included (28 girls and eight boys). Their age ranged from 3 to 42 months (mean 10.2±9.6 months). Besides the recurrent cases, we excluded another group with different causes: 13 boys (one had a colostomy performed at birth, one had anorectal stenosis, and 11 underwent a cutback or Y-V anoplasty) and nine girls (three underwent cutback anoplasty, three had presacral teratoma ‘Currarino syndrome’, two had incomplete cloaca ‘common urogenital sinus+rectoperineal fistula’, and one had caudal duplication syndrome).
In the study group (28 girls and eight boys), we found associated anomalies in three girls (10.7% of the female patients): congenital heart disease in one patient, an absent vagina in another, and vesicoureteric reflux and anterior sacral meningocele in one patient.
All patients in the study (36) underwent limited sagittal anorectoplasty without protecting colostomy. The overall incidence of postoperative wound dehiscence was 22.2% (eight patients), one of which was isolated midline dehiscence without the involvement of the mucocutaneous anastomosis (Fig. 4). The incidence of wound dehiscence was higher in girls (7/28) than in boys (1/8), but it did not reach statistical significance (P=0.4, Mann–Whitney test) (Table 1).
The patients in the study were divided into two groups according to the extent of rectal dissection and mobilization during the operation (Table 1). During the first half of the study (2011–2012), we performed limited dissection and mobilization of the rectum (about 2–3 cm) (Fig. 3a). In this group (15 patients), there was a high incidence of wound complications (six patients, 40%). Five patients were considered to have a major dehiscence (more than half the circumference of the anoplasty) and four required a rescue colostomy. Because of the relatively high incidence of wound complications in the first group, we shifted to more dissection and mobilization of the rectum (about 4–5 cm) in the second half of the study (2013–2014). We called this ‘extended anorectal mobilization,’ which is more or less similar to the degree of dissection for a rectovestibular fistula (Fig. 3b). In the second group (21 patients), two had postoperative wound complications (9.5%) that were managed conservatively. The first had isolated midline dehiscence (Fig. 4), and the other had minor dehiscence at the mucocutaneous anastomosis of the anoplasty (Fig. 5a). Extension of the rectal dissection and mobilization decreased the overall incidence of wound complications significantly in the second group (Mann–Whitney test, P=0.04).
Low-anorectal anomalies comprise different clinical variants in both sexes (anterior ectopic anus, covered anus, bucket-handle deformity, anal stenosis). Recently, a single term, the ‘rectoperineal fistula,’ has been proposed to cover most of these variants 10. Basically, it describes a rectum that has passed through the pelvic floor (levator muscle) successfully to end into the perineum anterior to the center of the muscle complex. Therefore, the bowel termination is located within most of the sphincter mechanism, and fecal continence is expected to be excellent in these patients 10. However, constipation constitutes a major problem among these patients, which is sometimes so intractable as to cause fecal soiling (pseudoincontinence) 4.
There is no consensus on the best management for rectoperineal fistula. Some scholars would deny any surgical correction, especially when the rectoperineal fistula is sufficiently wide (what they call an ‘anteriorly displaced anus’) 4. They build their argument on the reported high incidence of postoperative constipation that would suggest little expected benefit from surgery. Others advocate prolonged regular dilatation for managing a narrow fistula 4. Simple anoplasty (cutback) is one surgical option that has stood the test of time, with minimal risk of wound complications. However, it is recommended only for selected cases (those with minor anterior displacement); otherwise, it will leave a very short perineum, which may be cosmetically unacceptable, in addition to the persistence of vestibular contamination in girls 4,6. In contrast, the limited sagittal anorectoplasty offers full correction by mobilization of the anorectum to be repositioned backwards in the center of the muscle complex. However, there are reports on the increased risk of wound dehiscence, anal retraction, and stenosis. This may affect the continence potential in these patients, in addition to the possible need for fecal diversion (colostomy), which may be considered as a significant morbidity for such a minor form of the disease 4,8,9.
This report is concerned with patients with rectoperineal fistula (both male and female) who underwent a limited sagittal anorectoplasty primarily without a protecting colostomy. We preferred to call our technique a ‘sagittal anorectoplasty’ as it combines concepts from both the anterior and the posterior sagittal approaches. One reason for this study was that we noticed a relatively higher incidence of wound complications among these patients compared with those with rectovestibular fistula.
We classified wound complications into either isolated midline skin dehiscence or those involving the mucocutaneous anastomosis also. The first type occurred only in one case, which was managed conservatively and healed by secondary intension without complications. It appears that the dissection of the fistula from the perineum (especially when wide) results in a local tissue defect that would exert extra tension on the midline skin closure in patients with rectoperineal fistula (Fig. 4). In contrast, dehiscence involving the mucocutaneous anastomosis was more common (seven patients) and probably more important due to the risk of anal retraction and stenosis. This was further subclassified into minor and major dehiscence according to the degree of circumferential wound dehiscence. Major dehiscence involves more than half the circumference, and is associated with significant mucosal retraction. The latter is more liable to delayed complications (fibrosis and stenosis).
Many factors have been investigated to decrease wound complications after primary sagittal anorectoplasty: early repair in the neonatal period, protective colostomy, preoperative total bowel irrigation, therapeutic antibiotics, and delaying the postoperative oral intake with or without parenteral nutrition 8,14,15. However, we agree with Pena and colleagues, who highlighted the importance of a tension-free mucocutaneous anastomosis 9,11–13. Effective mobilization of the anorectum through the proper plane of dissection (that maintains the intramural blood supply) is considered as the most important step in the operation that can guard against wound complications 11. However, to our knowledge, it is not clear in the literature as to what exactly is considered as an ‘effective’ mobilization in cases of rectoperineal fistula. Some reports discussed details about the appropriate extent of dissection in girls with rectovestibular fistula: Levitt and Pena 16 recommended the extension of the dissection cephalad until encountering the areolar tissue separating the rectum from the vagina; Okada et al.14 reported the dissection of 4–5 cm of the rectum; others recommended reaching up to the cervix of the uterus 13. We noticed that there is always a tendency to perform less dissection with the rectoperineal than with the rectovestibular fistula, which appears to be proportionate to the minor degree of the former. However, in our series, this was successful only in 60% of the cases and was associated with wound complications in 40% of the cases. By extending the dissection of the rectoperineal fistula almost similar to a rectovestibular dissection, the incidence of postoperative wound complications decreased significantly to 9.5%.
The study may be criticized for comparing two groups that were not operated synchronously, and that other factors related to the growing experience of the surgeon might have influenced the improvement of the results. However, both groups were comparable (regarding age and sex), and all patients were operated by the same surgeon (with experience in pediatric surgery for 15 years), who performed the same surgical technique, except for the modification related to the extent of rectal mobilization in the second group. Also, the study did not address the delayed and functional outcomes of the procedure, which we thought to be studied and discussed in another separate report.
Among patients with rectoperineal fistula, extension of the dissection and mobilization of the rectum from the anterior structures (vagina in girls and bulbospongiosum in boys) decreases wound dehiscence after a limited sagittal anorectoplasty procedure.
Conflicts of interest
There are no conflicts of interest.
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