A case of primary transitional cell carcinoma of the male urethra is reported. A 24-year-old man, who complained of tumor of the urethral orifice with hypospadias, was admitted to our Hospital on November 30, 1983. Urethrocystoscopy and biopsy revealed grade 1 papillary transitional cell carcinoma of the anterior urethra. Partial resection of anterior urethra including tumor was performed on December 20, 1983. Histopathological diagnosis revealed grade 2 transitional cell carcinoma without submucosal invasion. Postoperatively, the patient was treated with Tegafur and Krestin as adjuvant immuno-chemotherapy. The patient has shown no evidence of disease for 3 years after surgery. There have been reported 23 cases of primary transitional cell carcinoma of the male urethra in Japan.