Abstract
Von Hippel-Lindau (VHL) disease can cause epididymal cystadenomas, typically asymptomatic. We report a 27-year-old male with a VHL history (resected CNS hemangioblastoma) presenting unusually with recurrent hemospermia and bilateral epididymal enlargement. Ultrasound and CT confirmed bilateral epididymal lesions and other VHL-associated findings (pancreatic/renal). Persistent hemospermia prompted bilateral epididymectomy, confirming papillary cystadenomas. This case highlights an atypical, symptomatic presentation of VHL-related epididymal cystadenoma requiring surgical intervention, contrasting with usual conservative management for these benign lesions.