Scholarly article on topic 'Post-Swenson's rectourethral fistula repair using the posterior sagittal route'

Post-Swenson's rectourethral fistula repair using the posterior sagittal route Academic research paper on "Clinical medicine"

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Abstract of research paper on Clinical medicine, author of scientific article — Michelle V. Vincent, Newton D. Duncan

Abstract Formation of rectourethral fistulas following pullthrough procedures for Hirschsprung's disease is a rare but recognized complication. Surgical correction is challenging, with use of vascularized grafts associated with high recurrence rates. We report on such a complication in a young boy whose repair was first approached anteriorly, utilizing vascularized omentum as a urethral patch. With recurrence of the fistula the urethral breech was ultimately repaired by direct tissue approximation via the posterior sagittal route.

Academic research paper on topic "Post-Swenson's rectourethral fistula repair using the posterior sagittal route"

J Ped Surg Case Reports 1 (2013) 136-138

journal of Pediatric Surgery

CASE REPORTS

ELSEVIER

Contents lists available at SciVerse ScienceDirect

Journal of Pediatric Surgery CASE REPORTS

journal homepage: www.jpscasereports.com

Post-Swenson's rectourethral fistula repair using the posterior sagittal route

Michelle V. Vincent*, Newton D. Duncan

The University Hospital of the West Indies, Department of Surgery, Anaesthesia and Intensive Care (Pediatric Surgery), Kingston 7, Mona, Jamaica

ARTICLE INFO ABSTRACT

Article history: Formation of rectourethral fistulas following pullthrough procedures for Hirschsprung's disease is a rare

Received 2 April 2013 but recognized complication. Surgical correction is challenging, with use of vascularized grafts associated

Received in revised form with high recurrence rates. We report on such a complication in a young boy whose repair was first

I7 April2013 approached anteriorly, utilizing vascularized omentum as a urethral patch. With recurrence of the fistula

Accepted 7 May 2013 , , , , , , . , ■ ■ ■ , ■ ■ ,

the urethral breech was ultimately repaired by direct tissue approximation via the posterior sagittal

route.

"T " © 2013 Elsevier Inc. All rights reserved.

Key words:

Hirschsprung's disease Rectourethral fistula Posterior sagittal approach

Formation of fistulas following pullthrough procedures for Hirschsprung's disease is a rare but recognized complication. It is thought to result as a further complication of anastomotic leaks or disruption. Spontaneous closure of these fistulas is rare and is best managed by closure using a combined abdominal and posterior sagittal approach.

1. Case report

An eight-year-old boy, weighing 20.4 kg (below the 50th centile) was referred to our pediatric surgical unit with a history of chronic constipation dating back to early infancy. This was associated with intermittent bouts of abdominal distension and laxative use. Fecal loading was apparent on abdominal examination and a digital rectal examination revealed normal sphincter tone with copious amounts of firm stool in the rectum. Daily rectal washouts were commenced and he underwent an open rectal biopsy, which revealed aganglionosis with associated neuronal hypertrophy, confirming the diagnosis of Hirschsprung's disease.

He underwent a primary Swenson pullthrough procedure using a combined anterior abdominal and perineal approach. At laparotomy performed via a hockey-stick incision the sigmoid colon and distal descending colon were noted to be markedly dilated. On completion of pelvic dissection via the abdominal incision which was not difficult, the perineal portion of the procedure was completed using a hand sewn, single-layered anastomosis. Mid-

* Corresponding author. Tel.: +876 866 9745; fax: +876 970 4302. E-mail address: michvincent@yahoo.com (M.V. Vincent).

2213-5766/$ — see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2013.05.002

sigmoid colon formed the proximal limb of the anastomosis based on frozen section analysis of serial seromuscular biopsies.

On day 2 postoperatively the child was noted to be intermittently pyrexial. This was not associated with peritonism, perianal edema/erythema and abated on day 3 postoperatively. He was thus discharged home on the 4th postoperative day.

Six days after his discharge the child represented with a five-day history of anorexia, fever and passage of malodorous watery diarrhea associated with one episode of vomiting on the day of presentation. On examination he was ill-looking, pyrexial and dehydrated with evidence of oropharyngeal thrush. He also had a painfully distended bladder. The child was resuscitated, started on broad-spectrum antibiotics, catheterized and had an abdominal ultrasound scan which excluded the presence of an intraabdominal collection. He was treated for postoperative/post-pullthrough hirschsprung's enterocolitis and twice daily rectal washouts were commenced. On day 4 of his admission whilst undergoing a rectal washout he was noted to have a feculent discharge per urethra, and it was at this time that the diagnosis of a rectourethral fistula was made. On exploratory laparotomy he was noted to have an approximate 50% anastomotic dehiscence and a defunctioning transverse colon double-barrel colostomy was thus performed, together with a suprapubic cystostotomy.

A distal colostogram performed postoperatively confirmed the presence of a fistulous tract between the neorectum (at the area of the disrupted anastomosis) and the posterior urethra, with rapid retrograde filling of the bladder noted during the procedure (Fig. 1).

Eight weeks after the diagnosis of a rectourethral fistula, when his nutritional status was optimized he underwent exploratory laparotomy, cystoscopy, fistula repair with an omental patch

Fig. 1. Patient's distal colostogram showing rapid retrograde filling of the bladder during the procedure.

overlay and a re-do Swenson procedure as definitive management of his rectourethral fistula. At cystoscopy, the fistulous opening was demonstrated just to the right of the verumontanum, through which a stent passed with ease into the pelvic cavity. The fistulous opening in the pelvic cavity was repaired using interrupted vicryl sutures. An omental patch was then mobilized down into the pelvis and oversewn over the repaired fistula. The distal end of the neo-rectum was then resected to healthy bowel, mobilized to ensure it was not under tension and then pulled through the anal orifice posterior to the omental patch. A single-layered anastomosis of this end of the bowel was then performed just proximal to the dentate line. The defunctioning transverse colostomy and suprapubic cys-tostomy were left in-situ.

Twelve weeks after re-do pullthrough and fistula closure an MCUG (micturating cystourethrogram) and distal colostogram, done at separate settings, did not demonstrate any persistent communication between the urinary and gastrointestinal tracts. He was thus taken back to theater and had closure of his colostomy, with removal of his suprapubic catheter. He represented however with urosepsis, with an MCUG (micturating cystourethrogram) showing a large colourethral fistula and had to have another colostomy performed. At this point he had a distal colon double-barreled co-lostomy performed and a suprapubic catheter was re-inserted.

One year later he was investigated with a distal colostogram performed using water-soluble contrast. The contrast was instilled into the distal limb of the stoma until the bowel was maximally distended. No colourethral fistula was demonstrated at this point and as a result his colostomy was closed. However it was soon apparent that spontaneous closure of the fistula had not occurred, and that the radiological studies performed were false-negative, when he again represented with abdominal distension, fever and passage of malodorous diarrhea within a few weeks of colostomy closure. Urine cultures were also supportive of this diagnosis, having grown a heavy growth of Escherichia coli. Re-do colostomy and suprapubic cystostomy were thus performed.

Definitive closure of the colourethral fistula was ultimately undertaken utilizing the combined abdominal and posterior sagittal approach, 30 months following his initial Swenson pullthrough procedure. First with the patient supine a laparotomy was performed. The colon was mobilized and divided very low in the pelvis. The distal end was sutured closed with sutures intentionally left

long and pushed deep down into the pelvis. During this procedure an appendicectomy and loop colostomy (mid-transverse colon) was also performed, the latter because of the atypical location of the appendix after this third pullthrough procedure. After closure of the abdomen the patient was then placed prone and a midline posterior sagittal incision was made without dividing the ring of the anus. The rectourethral fistula along with the pulled through colon (with long sutures attached) were clearly visible after longitudinally opening the posterior wall of the amputated pelvic colon. The fistula in the anterior colonic wall was closed en-masse using four interrupted 3—0 vicyl sutures. The colon advanced low in the pelvis at lapa-rotomy was then pulled down with the aid of the attached sutures and sutured to the anal verge below the site of fistula repair. The posterior sagittal incision was then carefully closed approximating adjacent muscle layers. Closure of his colostomy and suprapubic cystostomy was performed 5 weeks later after an MCUG (micturating cystourethrogram) (Fig. 2), and distal loopogram showed no communication between the urinary and gastrointestinal tracts.

The child has since had no symptoms of a recurrent fistula, even after his fecal and urinary tracts were un-diverted. Now 1 year since the combined abdominal and posterior sagittal approach to closure of his recurrent rectourethral fistula he is thriving, with passage of stool 2 to 3 times per day. His sense of an urge to defecate has returned and episodes of soiling have been decreasing. He is now fully integrated back in school and participating in a bowel management regime as per the recommendations of Pena [1] for children with incontinence following surgery for Hirschsprung's disease.

2. Discussion

A fecal fistula developing after definitive surgery for Hirschsprung's disease is a rare but challenging complication. Fistula location can be quite varied, including: rectourinary/rectourethral [1—4], rectovaginal [1—3,5], rectocutaneous/perianal [1,3,6] and recto-ileal [1,3]. Fistulous complications have been reported to occur after all of the most commonly used pullthrough procedures for Hirschsprung's disease — Soave [1,5,6], Duhamel [1,4], and Swenson [1,2].

These fistulas occur as a further complication of anastomotic leaks or disruption following a pullthrough procedure [7]. An anastomotic leak itself is more likely to occur in the presence of the following: tension on the anastomosis, ischemia of the rectal cuff or the distal colonic segment, and incomplete anastomotic suturing and/or inadequate stapling [2,7].

Fig. 2. MCUG (micturating cystourethrogram) obtained 12 weeks following the combined abdominoposterior sagittal approach for closure of colourethral fistulas-bladder and urethra outline clearly with no obvious communication with the bowel.

Management of these fistulas is technically challenging and while successful attempts at conservative management have been described, managing them without performing a re-do procedure has proved unsuccessful in the majority of cases [1,3,8]. Operative management can be broadly categorized into 2 types of procedures:

1) Vascularized flap interpositions and

2) Re-do pullthrough procedures, utilizing a combined abdominal and posterior sagittal approach.

2.1. Vascularized tissue flaps

Vascularized tissue flaps were the original surgical procedures described for the management of post-pullthrough fistulas in patients with Hirschsprung's disease. It was modeled after techniques described in the adult literature for similar fistulas resulting from other conditions such as trauma, infection, inflammation, neoplasia, or iatrogenic injuries [4,9,10]. These included the use of tissue such as omentum, scrotal skin/dartos muscle [9,11], gracilis muscle [12,13] or rectal wall [10,14] interposed between the rectum and the urethra/vagina.

2.2. Posterior sagittal abdominoperineal pullthrough (PSAP)

The newer approach to the management of post-pullthrough fistulas for Hirschsprung's disease is a re-do pullthrough procedure using a combined abdominal and posterior sagittal approach. This surgical approach was initially used for the correction of ano-rectal malformations by deVries and Pena in the early 1980's, and later extended to include management of rectourethral fistulas [10,15,16].

When applied for use in children with fistulas, which developed post-pullthrough procedures it was heralded for its use in cases where pelvic dissection would be difficult. This was often noted because of dense fibrosis resulting from the previous pullthrough procedure itself, or more commonly from its complications -anastomotic leaks or dehiscence. Once fibrosis has developed identification and mobilization of the fistula can be extremely difficult [4,8]. The posterior sagittal approach obviated the need for difficult pelvic dissection. In addition large/broad fistulas could also be adequately managed using the posterior sagittal approach since the procedure offered excellent exposure, precise dissection, and the ability to perform a direct closure of the fistula [4,8,13].

Another major advantage to this newer approach is the fact that even if the fistula is not identified, re-fistulization does not occur because of the presence of the rectal sleeve brought down [13]. Finally, the presence of anorectal stenosis, which may complicate anastomotic leaks with fistula formation, is not a contraindication to use of this procedure [13].

In 1995 Georgeson [17] initially reported laparoscopic-assisted options for HD (Hirschsprung's disease), the biggest advantage being minimal invasiveness and superior visualization of vessels and the ureters. Presently laparoscopic-assisted techniques have now also been utilized with success for the combined abdominal and posterior sagittal approach to the management of rectourinary fistulas [13].

This challenging case has been a learning point for us in many respects. In addition only a few case reports have so far been published demonstrating the success of the combined abdominoperineal approach to the management of rectourethral/ rectovaginal fistulas in children with Hirschsprung's disease [2,4,8,13]. We thus thought it important to share this case with colleagues. Firstly, in retrospect, this child may have better been served by an initial leveling colostomy prior to having a pullthrough procedure, after his delayed diagnosis of Hirschsprung's disease at the age of 8 years. This would have allowed the associated mega-colon to resolve prior to the pullthrough procedure, thus possibly

markedly decreasing the risk of anastomotic dehiscence with subsequent formation of a rectourethral fistula.

Secondly, spontaneous closure of rectourethral fistulas is rare, and radiological investigations may not always adequately demonstrate the presence of a fistula. We proceeded to close this patient's colostomy approximately 18 months after a recurrent rectourethral fistula was demonstrated following the omental patch onlay procedure. This decision was based on the child's improved nutritional state, length of time elapsed (18 months) and the result of a distal colostogram demonstrating no persistent fistula, all of which lead us to erroneously believe that the fistula had healed spontaneously.

3. Conclusion

Though use of vascularized flaps were the original surgical procedures used for closure of rectourethral fistulas, there is an increasing body of evidence that use of a combined abdominal and posterior sagittal approach is associated with superior cure rates [2,4,8,13]. This case adds to the few published series demonstrating the success of this approach.

Conflict of interest

As the corresponding author, I wish to confirm that none of the authors have any conflict of interest to declare.

Sources of funding

As the corresponding author, I wish to confirm that none of the authors have any sources of funding to declare.

References

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