Lichen sclerosus (LS) is a rare disease with malignant potential and has been identified by the European Society of Urology Guidelines as a risk factor for penile squamous cell carcinoma (SCC) cancer. LS combined with urethral squamous cell carcinoma (USCC) is extremely rare. There has been only one case report of this combination in China over the last decade. The prevalence of this combination in East Asian population is unclear. In this report, a 49-year-old patient with the presence of LS, perineal masses, and abscesses was hospitalized. He underwent anti-infectious therapy, meatotomy, perineal abscess incision, and drainage and debridement of the perineal wound. USCC was diagnosed following a series of biopsies and treated with radiation therapy. The patient remains well and alive with no recurrence of USCC 13 months after radiation treatment.