The incidence of osteomyelitis has dramatically declined since the introduction of antibiotics. When it now occurs in western society, the possibility of predisposing immunosuppressive conditions or underlying bony disease, such as Paget's disease or osteopetrosis, should be considered. Osteopetrosis (marble bone disease, Albers-Schonberg disease), is a congenital sclerosing disease of bone characterized by osteoclastic dysfunction. There is normal production of bone with lack of physiologic resorption. Bone involved with osteopetrosis is thought to have a compromised vascular supply. Local infection such as odontogenic infection is more likely to lead to osteomyelitis, which is a complication in 10% of reported cases of osteopetrosis.(1) These cases have occurred almost exclusively in the mandible. Osteomyelitis of the maxilla is very rare, probably because of the thin cortical bone and rich collateral blood supply. This article reports a case of aggressive unresolved osteomyelitis of the maxilla in an adult patient.