Abstract
Urethrocutaneous fistula is a rare complication of circumcision, especially after non-medical procedures. We report a 4-year-old boy with a long-segment post-circumcision urethrocutaneous fistula following traditional circumcision at age one. He presented with complete urinary diversion via a ventral penile opening, with a preserved orthotopic meatus and well-formed urethral plate. He underwent Duplay-type tubularization without urethral plate incision, reinforced with a dartos flap. Recovery was uneventful, with normal voiding and no recurrence at one year. This case shows that preservation of the urethral plate allows successful single-stage reconstruction with good functional and cosmetic outcomes.
Keywords: Urethrocutaneous fistula, Circumcision complications, Pediatric urology, Urethral reconstruction, Duplay technique, Dartos flap
1. Introduction
Circumcision is one of the most commonly performed surgical procedures worldwide and is generally safe when carried out in appropriate medical settings. However, complications still occur, particularly when the procedure is performed by untrained practitioners or under suboptimal conditions.1
Among these, urethrocutaneous fistula is an uncommon but particularly distressing complication, often resulting in both functional urinary disturbance and unsatisfactory cosmetic outcomes.2 Most post-circumcision fistulas are small and localized, while long-segment defects are rare and more challenging due to the extent of urethral involvement.3
Surgical management is guided by principles derived from hypospadias repair, including preservation of the urethral plate when feasible, tension-free tubularization, multilayered closure, and vascularized tissue interposition to reduce recurrence.4
A normal orthotopic meatus together with a well-formed urethral plate in a long-segment defect is unusual and provides a favorable anatomical basis for reconstruction using the Duplay principle.5 This avoids more complex substitution techniques and allows anatomical repair.
Such cases remain rarely reported, and optimal management is not well defined.6,7
We report a rare case of this condition managed successfully using Duplay-type tubularization reinforced with a dartos fascia flap, with excellent functional and cosmetic results.
2. Case presentation
A 4-year-old boy was referred with an abnormal urinary stream following circumcision performed at the age of one year by a non-medical traditional circumciser. Since the procedure, he had been passing urine entirely through a ventral opening along the penile shaft. He was urinating normally before circumcision. There was no history of congenital penile anomaly, urinary tract infection, or prior urological surgery other than circumcision.
On physical examination, the native urethral meatus was orthotopic and located at the tip of the glans. A long ventral urethrocutaneous fistulous tract was identified, extending from the subcoronal region to the proximal penile shaft. The intervening urethral plate was well-formed, supple, mobile, and of adequate width to permit tension-free tubularization, with no penile curvature. The remainder of the external genitalia was normal (Fig. 1).
Fig. 1.
Preoperative view showing long ventral urethrocutaneous fistula with preserved orthotopic meatus.
These findings were consistent with a long-segment post-circumcision urethrocutaneous fistula with preservation of the distal urethral meatus and an intact urethral plate.
3. Management and surgical technique
The patient underwent surgical repair under general anesthesia. A stay suture was inserted through the glans penis for traction. A U-shaped incision was fashioned around the urethral plate, followed by complete penile degloving. The urethral plate was mobilized bilaterally off the corpora to ensure tension-free tubularization.
Given the favorable characteristics of the urethral plate, tubularization was performed without midline incision (Duplay technique). The plate was tubularized over a 6 Fr Foley catheter using 5-0 polyglactin 910 sutures in two layers. The first layer consisted of a continuous subepithelial layer, followed by a second reinforcing interrupted layer (Fig. 2).
Fig. 2.
Intraoperative tubularization of the urethral plate using Duplay technique without midline incision.
A well-vascularized ventral dartos pedicle was harvested bilaterally and transposed to cover the neourethra as an additional protective layer, in line with established principles for reducing fistula recurrence.7 Skin closure was completed in a tension-free manner. A urethral catheter was left in situ for 10 days.
4. Outcome
The postoperative course was uneventful. After catheter removal, the patient voided with a normal-caliber urinary stream without leakage (Fig. 3).
Fig. 3.
Patient urinating normally from the tip of the glans penis 10 days after transurethral catheter removal.
At one-year follow-up, he remained asymptomatic with satisfactory functional and cosmetic outcomes and no evidence of fistula recurrence.
5. Discussion
Post-circumcision urethrocutaneous fistula is an uncommon complication, most frequently reported following procedures performed outside standard medical settings.1 While most cases involve small, localized fistulas, long-segment urethral defects are rare and represent a more complex reconstructive challenge due to the extent of urethral involvement and the need to preserve urethral continuity.2
In previously published literature, fistula repair is more commonly described following hypospadias surgery, where tissue quality may be compromised.3 In contrast, post-circumcision injuries may preserve relatively healthy urethral plate tissue, allowing anatomical reconstruction.
Reconstruction is frequently based on hypospadias principles, particularly tubularized incised plate urethroplasty (TIP), which is widely used for narrow or deficient urethral plates.4 However, TIP requires a midline incision, which may introduce fibrosis and carries a risk of stricture formation.
In the present case, the urethral plate was wide, supple, and well-vascularized, allowing Duplay-type tubularization without incision, thereby preserving native tissue integrity. Dartos fascia interposition provided an additional protective layer and is well established in reducing fistula recurrence.5
The decision to utilize the Duplay principle over the TIP technique was based on the specific pathophysiology of this injury. Unlike congenital hypospadias where the urethral plate may be dysplastic, post-circumcision trauma often leaves behind healthy, well-vascularized tissue. By avoiding a midline relaxing incision, structural integrity was maintained and the risk of fibrosis or stricture minimized.
The combination of a long-segment defect with preservation of both the distal meatus and a well-formed urethral plate is rarely reported. This case supports an anatomy-guided approach where urethral plate quality and vascularity may be more decisive than defect length in selecting reconstruction strategy.6
Importantly, this reinforces individualized surgical planning. In selected patients, preservation of a healthy urethral plate and avoidance of midline incision may reduce fibrosis while still enabling a tension-free reconstruction 7, 8, 9.
This report is limited by its single-case design, and further studies are required to validate reproducibility.
6. Conclusion
Long-segment post-circumcision urethrocutaneous fistula is rare and challenging. This case shows that when the urethral plate is preserved, wide, and well-vascularized, Duplay-type tubularization without incision, reinforced with dartos fascia, can achieve excellent functional and cosmetic outcomes.
ICMJE statement
All authors meet authorship criteria.
CRediT authorship contribution statement
Zubair Mohamed Dahir Sed: Writing – review & editing, Writing – original draft, Supervision, Methodology, Data curation, Conceptualization. Badrudiin A. Warsame: Writing – review & editing, Data curation.
Consent for publication
Written informed consent was obtained from the patient's legal guardian for publication of this case report and accompanying clinical images. A copy of the consent is available for editorial review upon request.
Guarantor
Dr. Zubair Mohamed Dahir Sed accepts full responsibility for the integrity of the work as a whole, from inception to publication.
Ethical approval
Ethical approval was not required for this single case report according to institutional policy at Shaafi Hospital, Mogadishu, Somalia.
Funding
None.
Conflict of interest
None.
References
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