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Craniofacial deformity and malocclusion are primary concerns following temporomandibular joint ankylosis (TMJa) in growing patients, and they pose even greater challenges in adult patients. The treatment objectives always involve restoring proper jawbone structure, achieving stable occlusion, and attaining satisfactory joint mobility. This report presents a 4-year follow-up of an adult patient with TMJa-induced mandibular deviation, who underwent a combined treatment approach involving distraction osteogenesis (DO) and orthodontic–orthognathic surgery. Orthodontic treatment resulted in favorable occlusion and improved facial esthetics. A new condyle with a reconstructed glenoid fossa in a forward position was established after mandibular DO and the damaged TMJ experienced self-remodeling owing to functional improvement. Thus, this case demonstrates the efficacy of DO in promoting adaptive TMJ self-remodeling with long-term stability when treating mandibular deviation caused by condylar ankylosis in adult patients.