Malignant tumours of the male urethra are rare and commonly present at an advanced stage. The most common type of urethral cancer is urothelial carcinoma, followed by squamous cell carcinoma. Less than 5% of urethral cancers are adenocarcinoma. We report a young male with signet ring cell carcinoma of the membranous urethra who presented with recurrent lower urinary tract symptoms. Although he was initially treated for a urinary tract infection, the correct diagnosis was made with an appropriate imaging workup. He underwent radical urethrectomy with total pelvic exenteration with bilateral extended pelvic lymph node dissection. Ileal conduit and colostomy were performed as urinary and bowel diversions, respectively. The patient received adjuvant chemotherapy, however, succumbed to COVID-19 8 months after the procedure. This case has been presented to highlight the high index of suspicion required to diagnose this rare malignancy.
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