Scholarly article on topic 'Pneumaturia signaling a fistula between the rectum anastomosis and seminal vesicle as a complication after transanal endorectal pull-through operation for Hirschsprung's disease. A method of repair'

Pneumaturia signaling a fistula between the rectum anastomosis and seminal vesicle as a complication after transanal endorectal pull-through operation for Hirschsprung's disease. A method of repair Academic research paper on "Clinical medicine"

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{"Hirschsprung's disease" / "Transanal endorectal pull-through" / Fistula / "Seminal vesicle" / Rectum / "Transperineal approach"}

Abstract of research paper on Clinical medicine, author of scientific article — Christina Granéli, Pernilla Stenström, Anna Börjesson, Antoni Zawadzki, Hans Frederiksen, et al.

Abstract A 2-year-old boy underwent an uneventful transanal endorectal pull-through for Hirschsprung's disease. Postoperatively he suffered from pneumaturia which prompted surgical evaluation. He was found to have a rectum to seminal vesicle fistula. He was re-operated closing the fistula through an anterior transperineal approach with a successful operative outcome.

Academic research paper on topic "Pneumaturia signaling a fistula between the rectum anastomosis and seminal vesicle as a complication after transanal endorectal pull-through operation for Hirschsprung's disease. A method of repair"

journal of Pediatric Surgery

CASE REPORTS

J Ped Surg Case Reports 2 (2014) 439-441

Contents lists available at ScienceDirect

Journal of Pediatric Surgery CASE REPORTS

journal homepage: www.jpscasereports.com

Pneumaturia signaling a fistula between the rectum anastomosis and seminal vesicle as a complication after transanal endorectal pull-through operation for Hirschsprung's disease. A method of repairq

Christina Granelia,d'*, Pernilla Stenströma,d, Anna Börjessona,d, Antoni Zawadzkib, Hans Frederiksenc,d, Magnus Anderberga,d, Einar Arnbjörnssona,d

a Department of Paediatric Surgery, Skäne University Hospital, Lund, Sweden b Department of Surgery and Skäne University Hospital, Lund, Sweden cDepartment of Urology, Skäne University Hospital, Malmo, Sweden d Institution of Clinical Research, Lund University Lund, Sweden

ARTICLE INFO ABSTRACT

Article history: A 2-year-old boy underwent an uneventful transanal endorectal pull-through for Hirschsprung's disease.

Received 2 August 2014 Postoperatively he suffered from pneumaturia which prompted surgical evaluation. He was found to

Received in revised form have a rectum to seminal vesicle fistula. He was re-operated closing the fistula through an anterior

25 to^t2014 transperineal approach with a successful operative outcome.

Accepted 25 August 2014 K . , ■ , .„ ■ ,

© 2014 The Authors. Published by Elsevier Inc. All rights reserved.

Key words:

Hirschsprung's disease Transanal endorectal pull-through Fistula

Seminal vesicle Rectum

Transperineal approach

In some children with Hirschsprung's disease (HD) undergoing transanal endorectal pull-through (TERPT), the initial surgical procedure fails, and the child suffers from repeated or persistent symptoms. These children present with severe inflammation, intestinal obstruction, faecal or urinary incontinence or abdominal pain often requiring operative intervention [1—10]. Here we report on a boy who was discovered to have a fistula between the rectal anastomosis and his right seminal vesicle postoperatively after a transanal endorectal pull-through for Hirschsprung's disease with a transition zone in the middle of the sigmoid colon.

q This is an open access article under the CC BY-NC-ND license (http:// creativecommons.org/licenses/by-nc-nd/3.0/).

* Corresponding author. Institution of Clinical Research, Lund University, Lund 22185, Sweden. Tel.: +46 702986991.

E-mail address: christina.graneli@med.lu.se (C. Granéli).

1. Case report

A boy aged 23 months was diagnosed with Hirschsprung's disease (HD) after problems with severe constipation. The diagnosis was suspected when the recto-anal inhibitory reflex could not be visualized with a modified contrast enema [11] disclosing a transition zone in the sigmoid colon. The diagnosis was verified by rectal suction and full wall biopsy according to the routine procedure at that time [12].

The boy underwent a routine TERPT operation [13,14], similar to the Swenson procedure, at the age of two years. The operating surgeon reported a standard operating procedure. The boy returned home on the third post-operative day after an uneventful postoperative course.

On returning home the boy presented with urinary tract symptoms which were interpreted as a urinary tract infection and treated with antibiotics. Urine culture disclosed no bacteriuria. The

2213-5766/$ — see front matter © 2014 The Authors. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2014.08.012

boy's guardian noted that the boy had pneumaturia at every micturition. This prompted surgical evaluation.

Repeated X-ray examinations of the urinary tract by a voiding cystourethrogram (VCUG) did not demonstrate any signs of a fistula to the rectum. After two normal X-ray examinations of the rectum and colon, an anography finally disclosed a part of a fistula from the anastomosis after the TERPT operation. The fistula was located in the TERPT anastomosis, 1.5 cm above the dentate line in the midline on the anterior wall of the rectum. The contrast used for performing the X-ray examination, Omnipaque®, did not pass to the urinary tract, thus not fulfilling the radiological definition of a fistula.

Examination under general anesthesia revealed a fistula opening in the scar after the TERPT operation, localized in the midline on the anterior wall of the rectum. The X-ray finding was thus verified. An excision of the fistula, undermining the bowel wall and closing it from the rectum did not lead to cessation of the pneumaturia indicating that fistulation was still present.

Urethrocystoscopy did not show any fistula from the urethra or from the urinary bladder. The openings of the ejaculatory ducts were visualized. A guide wire which was gently slid through the opening of the right ejaculatory duct appeared in the rectum through the opening described above. Thus, the child's fistula was discovered within a few days postoperatively. The symptoms were at first interpreted as a urinary tract infection causing some delay in the exact diagnosis.

The boy underwent a laparoscopy-assisted divided sigmoidos-tomy. A diverting colostomy was considered necessary to optimize the postoperative healing process after the closing of the fistula. The fistula was identified using 3D anal ultrasound. A modified method for closing the fistula was planned. The fistula was closed through a perineal transverse incision anterior to the rectum (Fig. 1). The rectal wall was visualized in the operating wound and the fistula was closed from the outside (Fig. 2).

In the first attempt a guide wire was placed in the fistula through the right seminal vesicle orifice i.e. the urethral opening of the ejaculatory duct. The fistula was divided in the middle and sutured. A 4-layered Biodesign (CooK Medical) was placed between the fistula ends, in a manner similar to the LIFT procedure [15]. After the first attempt for surgical repair, the fistula seemed to persist. Therefore a new operation to close it was made.

At the second attempt we saw that the first fistula was closed, but that the patient had a second one that was not detectable on X-ray and we had failed to see it during the first operation. A guide wire was placed in the fistula through the right seminal vesicle orifice. The fistula was divided in its middle portion. The anal portion was then cored out in its entirety, and the defect in the sphincter was closed in multiple layers with sutures. The urethral portion of the fistula tract was excised. To avoid the risk of stricture formation and/or closing of the ejaculatory duct the defect was closed in several layers of tissue outside the urethra. The procedure for this particular patient was a "mixture" of core-out used in the advancement flap procedure [16—18], and closure of fistula that is described in the treatment of recto-vaginal fistulae with the transperineal approach.

The sigmoideostomy was closed six months later, after imaging, leaving the boy in an acceptable condition and not different from what was achieved after the original TERPT operation. He is not using oral laxatives regularly, does not suffer from constipation, is passing stools up to four times a day and does not have any soiling.

2. Discussion

Recto-urethral fistulae and fistula between the rectum and the seminal vesicle are absolutely unacceptable complications of transanal surgery and can be avoided by adherence to basic surgical

Fig. 1. The figure shows the approach through a transverse incision in the perineum anterior to the anus.

principles [19]. Nevertheless, these complications do indeed occur and have to be dealt with. The complication here described was caused by the dissection at the TERPT operation. This complication can be avoided by a careful and meticulous dissection only.

The surgical approach to these fistulae can be discussed. In the case described here we chose a minimally invasive approach through a transverse perineal incision anterior to the anus. This surgical intervention turned out to be successful and did not influence the child's bowel control compared to that already achieved after the initial TERPT operation.

Approach

Fig. 2. The figure demonstrates the approach to the rectum to seminal vesicle fistula in order to close the opening on the anterior wall of the rectum.

Alternative methods include re-do TERPT [13] or a posterior sagittal anorectoplasty (PSARP) [3,19]. These methods are natural alternatives since they are routine surgical methods used by colo-rectal pediatric surgeons. Both these methods would inflict considerably more trauma to the already operated part of the child's body, would put the postoperative results after the TERPT operation at risk, and probably influence the child's bowel control.

Certainly, there are details in the operative intervention that should be discussed. These among others include the type of sutures to use. In our patient we used resorbable sutures only, as in all pedi-atric colorectal surgery. Furthermore, to insert tissue between the rectum and the urethra is considered necessary. In the patient described here, local tissue was used in the last and successful operation. The use of a biodegradable material is also possible. Moreover, the timing of the operative intervention is of importance. In the present case the child's symptom demanded prompt surgical intervention. He gained relief from the pneumaturia after the sigmoideostomy operation. However, a life with a sigmoideostomy is not a good option for a small child and that period should be as short as possible.

To summarize, we have found that the method described here has been shown feasible in closing of a rectum to seminal vesicle fistula. We hereby submit the method, well-known to adult colo-rectal surgeons and urological surgeons for evaluation by other pediatric surgeons.

3. Conclusion

We have described a case of fistula formation between the rectal anastomosis and the right seminal vesicle after a TERPT operation for HD with pneumaturia as the only symptom. Correction was performed by limited access to the fistula opening in the TERPT anastomosis on the anterior wall of the rectum via a perineal approach anterior to the anus. The intention was to avoid deterioration of the postoperative clinical signs of the HD after TERPT by performing re-do TERPT or PSARP incision or laparoscopic interventions and to avoid further surgical trauma to the previously operated area.

Consent

Informed consent was obtained from the child's guardians for publication of this case report.

Conflict of interest statement

The authors have no conflict of interest to disclose.

Source of funding

Acknowledgment

The authors are indebted to Gillian Sjödahl, Lexis English for Writers, Lund, Sweden, for linguistic revision of the manuscript.

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