Genitourinary melanoma is rare and classically associated with a poor prognosis. We describe our experience with 10 patients with penile or urethral involvement. In addition, we present what is to our knowledge the largest reported series of melanoma of the scrotum (6 cases). We reviewed the records of 16 men who presented consecutively to our institution with genitourinary melanoma between 1962 and 2000. Clinical and pathological characteristics were assessed, including Breslow thickness, primary surgical intervention and clinical course. Of 10 patients with penile or urethral melanoma 1997 American Joint Committee on Cancer melanoma pathological stage was T1 (depth less than 0.75 mm) in 4, T2 (0.75 to 1.5 mm) in 3 and T3 (1.51 to 4 mm) in 3. Only 1 of 4 patients with clinically palpable inguinal nodes had inguinal metastases at lymphadenectomy (BILND) and 3 who underwent prophylactic superficial BILND had negative findings. In 7 patients with T1-2N0M0 disease there were no local recurrences after wide local excision (WLE) or partial penectomy at a median followup of 35 months. Six of 7 men were rendered disease-free. One patient died of melanoma that developed at a second primary site. The 3 patients with T3 tumors who underwent partial (2) or radical (1) penectomy with or without BILND died of disease (2) or had progression (1). In all patients with penile melanoma the 5-year actuarial disease specific and recurrence-free survival rates were 80% and 60%, respectively, at a median followup of 39 months (range 20 to 210). Six patients with scrotal melanoma were treated with WLE without local recurrences. Three of the 6 patients had palpable inguinal nodes, of whom 2 died after chemotherapy for unresectable disease and 1 died of other causes 51 months after negative BILND. The 3 men with clinically negative groins who did not undergo prophylactic BILND had distant (1) or regional (2) metastases and died of disease. In patients with scrotal melanoma the 5-year actuarial disease specific and recurrence-free survival rates were 33.3% and 33.3%, respectively, at a median followup of 36 months. Partial penectomy or WLE provided effective local control for low stage penile or urethral melanomas and all scrotal lesions. Patients showing clinically positive, proven metastasis died despite appropriate surgical procedures and multi-agent chemotherapy. Prophylactic modified inguinal lymphadenectomy should be considered in select patients with penile, scrotal and anterior urethral melanoma.