Melanoma in situ including glans penis and fossa navicularis is unique and represents a challenging dilemma since surgery should not be very aggressive. We present a case of melanoma in situ treated with a two-stage penile and urethral reconstructive surgery, with emphasis on functional and aesthetic results. At the first-stage surgery an anatomic glansectomy was perfomed, and combined preputial and full-thickness skin grafts were used to prepare the urethral plate and restore the aesthetic aspect of the corpora cavernosa. After 6 months, the distal urethra was repaired including the configuration of an orthotopic meatus and a neo-glans.