ABSTRACT
INTRODUCTION: Urethral amyloidosis is a rare, probably inflammatory condition usually presenting with hematuria and obstructive urinary symptoms, thus mimicking urethral malignancy. After histological confirmation of the diagnosis, treatment can be expectant or symptomatic. AIM. To report an unusual cause of urethrorrhagia occurring only during erection in an otherwise healthy man. METHODS. A 30-year-old man presented with a 5-month history of urethrorrhagia occurring only during erection, and with a painless palpable nodule in his penile urethra clearly visible on urethral US and magnetic resonance imaging, but not on urethroscopy. RESULTS. The patient underwent wide surgical excision of the urethral nodule and grafting of the urethral defect with a pedicled preputial flap. Histological examination revealed isolated amyloid of urethral corpus spongiosum. CONCLUSIONS. Isolated urethrorrhagia during erection and without urinary symptoms can be the presenting sign of urethral amyloidosis involving corpus spongiosum rather than the urethral lumen; in such cases, surgical exploration, wide urethral excision and grafting are mandatory.