Urethroplasty may involve the use of tissue substitution including buccal mucosa graft (BMG), penile fasciocutaneous flaps, or skin grafts. Stricture recurrence and fistula formation are some uncommon complications that can result from surgery. The development of squamous cell carcinoma (SCC) after BMG substitution urethroplasty is a new complication that we encountered that has not been described in the literature. We present the first reported case of a patient who developed SCC of the buccal mucosa graft used to reconstruct the urethra.