A 21 years old female patient reported with the complaint of face asymmetry, pain, acne and rash on the skin in the mental area of the lower jaw (right side) and became sick at the age 1.5 years old after face trauma. She has experienced 4 low-efficient operations for diagnosis correction. Preoperative examination by 3D CT showed sclerosing focies in mental area of the mandible. On the base of anamnesis, clinical signs (pain, enlarged mandibular bone tissue, skin diseases, and typical radiological picture) were strongly suspected Garre osteomyelitis associated with SAPHO syndrome. Subsequent computer simulation (stereolithographic model, 3D-printed) allowed planning surgery. In January 2018 was held operation with extraoral approach–partial resection and leveling of the sclerosed bone in the area of lesion (according to printed CT scan model), dissection of soft tissue scars after previously performed operations. In 5 days, the patient left hospital in good condition with recommendations for further examination, sanation of odontogenic infection, radiological control. This disease refers to atypical primary chronic forms of osteomyelitis and is rare in clinical practice of maxillofacial surgeons, especially in adults. Treatment consists of surgical and conservative phases. The prognosis is favorable, but syndrome is often persistent.