PURPOSE: We report the case of a 69-year-old man who had suffered a third-degree burn injury of the buttocks close to the perianal region at the age of 2 years. Although initial attempts for conservative treatments failed, wound healing was achieved after numerous skin grafts. However, after prolonged healing, the patient developed scar contracture, resulting in total effacement of the gluteal folds and natal crease, and formation of a gluteal pseudoampulla and a pseudoanus. As a result of misinterpretation of the local anatomy, for the next 50 years the patient had to defecate into the gluteal pouch and empty this pseudoampulla by manually compressing the buttocks and irrigating with chamomile tea. METHOD: After a thorough examination revealed the presence of a normal, uninjured anus hidden by the scarred pouch, we excised this burn scar to expose the unburned perianal tissue and covered the resulting defect with a meshed, split-thickness skin graft. RESULT. Anorectal function was preserved, the anatomic contour was restored, and primary wound healing was achieved after one single-stage operation. CONCLUSION: To prevent such de formities, we recommend early excision and skin grafts as initial treatment, as opposed to conservative treatment, and thorough follow-up examinations to avoid misinterpretation of such a complex, altered local anatomy.