Squamous cell carcinoma of the urethra is a rare urologic malignancy. In spite of aggressive management with radical and often times disfiguring surgery and/or radiation therapy, prognosis remains poor. Initial success in treating squamous cell carcinoma of the esophagus and anal canal has been reported with a combined radiation and chemotherapy protocol. In hopes for improving the treatment outcome for patients with squamous cell carcinoma of the urethra, we have applied the new combination chemotherapy/radiation therapy protocol. Herein, we report the successful downstaging (clinical Stage C to pathologic Stage TO) and treatment of squamous cell carcinoma of the anterior urethra with combination chemotherapy/radiation therapy. This was followed with penile-preserving surgery to document local control of disease and to avoid the morbidity of a radical disfiguring operation.