Primary urethral cancer is a rare condition often manifesting in the sixth decade of life and is typically associated with urethral stricture disease. A 65-year-old male, a known case of stricture urethra, underwent perineal urethrostomy 10 years back. He presented with a growth at the urethrostomy site and the biopsy revealed squamous cell carcinoma. Magnetic resonance imaging of the pelvis revealed that the lesion extended into the penile and prostatic urethra with possible involvement of the corpus spongiosum, bulbospongiosus, and the bladder neck. Radical cystectomy, penectomy, scrotectomy, and bilateral orchiectomy with ileal conduit were performed. At 18-months of follow-up, the patient is recurrence-free.
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