A 58-year-old Caucasian male presented to the urology clinic reporting an approximate one-year history of a persistent irritating, slowly progressive, glans penis redness. Biopsy revealed penile squamous cell carcinoma in situ. He underwent a partial glansectomy with circumcision and skin grafting. At three months follow-up there is no evidence of local disease recurrence. In western countries, primary malignant penile cancer is uncommon, with an incidence of less than 1 per 100,000 males. Squamous cell cancer accounts for more than 95% of cases of penile cancer. Squamous cell carcinoma in situ on the penile mucosa or transitional surfaces is also known as Erythroplasia of Queyrat. In the region, one third of penile squamous cell carcinoma in situ cases progress to invasive squamous cell carcinoma.