Osteopetrosis represents a heterogeneous group of rare, hereditary bone disorders with variable clinical features, and an increase in bone density. Osteomyelitis of the jaws is a significant complication of osteopetrosis. In this article, a reported patient with osteopetrosis complicated by osteomyelitis of the mandible was examined. The patient was treated with intravenous antibiotic therapy, debridement of necrotic bone and hyperbaric oxygen therapy; in addition, the authors attempted to implant the calcium sulfate and vancomycin to reconstruct the bone defect. The patient demonstrated satisfactory healing, and no recurrence of osteomyelitis was observed during the 6-month follow-up period. The treatment of osteopetrosis complicated by osteomyelitis of the mandible is difficult. The treatment of osteopetrosis complicated by osteomyelitis is controversial. The authors recommend the following sequential treatment of osteopetrosis complicated by osteomyelitis of the mandible: systemic antibiotic therapy and hyperbaric oxygen therapy before and after surgery; debridement of the necrotic bone; sufficient periosteal coverage and adequate soft tissue to cover the wound; implantation with calcium sulfate and vancomycin to reconstruct the bone defect as much as possible, which may be helpful in treating the disease.
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