Urolithiasis is a well-known disease. Stones can form in all parts of the urinary tract. Diagnosis and treatment usually do not create clinical problems. However from time to time in daily practice doctors are confronted with casuistic cases. Here, we report a 54-year-old man with a large stone localized to the urethra. Because of the size of the stone, confirmed on radiography, and the presence of a cutaneous fistula, open surgery was performed. Due to urethral stricture, the patient required a second stage of reconstructive surgery. This case emphasizes that unusual presentation of urolithiasis may occur, and different surgical approaches as well as other surgical manipulations in such cases must be considered.