Orthognathic surgery in patients with craniofacial osteopetrosis, a condition associated with osteoclast dysfunction, is usually avoided because of the risk of osteomyelitis. A 19-year-old woman presented with the chief complaint of severe malocclusion and anterior crossbite. After radiographic evaluation, craniofacial osteopetrosis was diagnosed. Surgical-orthodontic treatment was performed after meticulous history taking and verification of normal bone turnover using bone-metabolism markers for endocrine evaluation. Favorable esthetic and functional outcomes were achieved. (Angle Orthod. 2021;91:555–563).
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