Glassy cell carcinoma is a poorly differentiated form of adenosquamous carcinoma that has never been reported in the urinary tract. We present the first case of primary glassy cell carcinoma of the urethra in a 48-year-old woman. She presented with a newly developed bulky mass protruding from her urethra. A biopsy of this mass revealed sheets of large polygonal cells with a "ground-glass" cytoplasm among a heavy inflammatory infiltrate, establishing the diagnosis of glassy cell carcinoma of the urethra. Treatment of her tumor included a combined surgical and chemotherapeutic approach.