Primary penile urethral cancer is a rare genitourinary malignancy arising from the epithelial lining of the urethra. Our patient is a 63-year-old male with newly diagnosed penile urethral carcinoma who presented with headache and was found to have leptomeningeal disease on imaging and cerebral spinal fluid analysis. He was treated with systemic and intrathecal chemotherapy with some response and improvement in symptoms. This is the first reported case of leptomeningeal disease as a complication of penile urethral carcinoma. Recognition and prompt treatment are important; however, overall prognosis of this entity remains poor.