ABSTRACT Introduction Persistent perineal wetness in adults with normal standard imaging is uncommon and may indicate rare congenital anomalies such as urethral duplication. Case Presentation A man in his early 20s presented with a 3‐year history of intermittent perineal wetness of unknown origin. Extensive gastrointestinal and urological investigations were unrevealing. Indigo carmine administration confirmed urinary leakage, prompting focused evaluation. Endoscopic assessment with meticulous perineal inspection under general anesthesia identified a tiny, non‐inflamed perineal opening. Fistulography and cystoscopy demonstrated an accessory tract arising from the prostatic urethra and opening to the perineum, consistent with Effmann type IIA2 Y‐type urethral duplication, extremely rare in adult‐onset. Complete excision was achieved using a combined perineal and laparoscopic transabdominal approach with light‐emitting catheter guidance, preserving urinary continence and erectile function. Symptoms resolved immediately without recurrence. Conclusion This case highlighted key diagnostic pitfalls and demonstrates effective surgical management of adult‐onset type IIA2 Y‐type urethral duplication.
Ueda et al. (2026) studied this question.