Introduction: The aim of this study was to evaluate the clinical features and prognosis of congenital anterior urethrocutaneous fistula (CAUF) patients. Methods: This is a retrospective chart review of patients diagnosed with CAUF and underwent surgical repair by the author between October 2017 and December 2021 that was performed at a single tertiary care institution. The medical records of CAUF patients were reviewed to evaluate their clinical characteristics and outcomes. Results: 11 patients with CAUF were included. The median age of the patients was 41 months (11–84 months). Subcoronal fistulas were observed in 1 patient, mid-penile shafts in 7 patients, proximal penile to subcoronal fistulas in 2 patients, and perineum to penile fistulas in 1 patient. Associated urinary system anomalies were found in 3 patients, and other anomalies were found in 2 patients. One-stage surgery was performed in 10 patients, and staged surgery was performed in 1 patient. There were no postoperative complications, and there was good voiding. Conclusion: Although CAUF often coexists with genitourinary or other deformities, it has a good prognosis following an appropriate surgical strategy for each patient.

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