The combination of Mohs micrographic surgery and sentinel lymph node biopsy in the treatment of penile melanoma is novel. Mohs surgery allows the removal of penile malignancies with microscopically controlled tumor-free borders, while maintaining cosmetic and functional demands through the maximal preservation of normal tissue. Sentinel lymph node biopsy minimizes the morbidity associated with inguinal node dissection. At 30 months follow-up, these two modalities together have achieved local control and regional nodal staging while minimizing functional morbidity.