Giant urethrocutaneous fistula following neonatal circumcision: a case report

Introduction and importance: Urethrocutaneous fistula is a recognized but uncommon complication of male circumcision. While most fistulas are small, giant urethrocutaneous fistulas associated with complete urinary diversion are exceedingly rare and present significant functional and reconstructive challenges. Presentation of the case: A 3-year-old male presented with complete urinary diversion through an abnormal ventral penile opening since neonatal circumcision performed by a traditional circumciser. The abnormal opening was first noted on the second postoperative day. Examination revealed a giant urethrocutaneous fistula measuring approximately 3 × 2.5 cm at the coronal sulcus, with a non-functional but non-stenotic distal urethral meatus, which was easily calibrated with an 8 Fr nasogastric tube. The patient underwent single-stage repair with two-layer urethral closure over an 8 Fr catheter, distal glans undermining, and vascularized dartos flap interposition. Postoperative recovery was uneventful. At 1 year follow-up, the child voided with a single forward urinary stream without recurrence or cosmetic deformity. Clinical discussion: Male circumcision is common and generally safe. However, complications can occur, including urethrocutaneous fistula, a rare but serious outcome, especially in neonates or when performed by untrained providers. Most fistulas are small and present with dual urinary streams. In contrast, giant fistulas with near-complete urinary diversion are extremely rare and difficult to repair. These injuries may result from urethral trauma, devascularization, or infection. Optimal management remains uncertain. We report a giant circumcision-related urethrocutaneous fistula successfully treated with single-stage multilayer urethroplasty reinforced by a dartos flap interposition. Conclusion: Giant urethrocutaneous fistula is a rare but severe iatrogenic complication of circumcision. In selected cases, single-stage multilayer repair, reinforced with dartos flap interposition, can achieve excellent functional and cosmetic outcomes.