Refractory maxillary osteomyelitis with osteopetrosis: A case report

In osteopetrosis, refractory jawbone osteomyelitis often occurs because of the progression of bone sclerosis, which makes the bone brittle. Herein, we report a case of refractory osteopetrosis-related maxillary osteomyelitis for which conservative therapy and surgical intervention were performed along with imaging evaluation. A 58year-old woman had maxillary gingivitis for 2 years, for which her tooth was recently extracted. Subsequently, she developed sequestration and oroantral fistula and visited our department. Panoramic radiography, computed tomography, and magnetic resonance imaging (MRI) scans revealed left maxillary osteomyelitis, sinusitis, and sequestration. She had been diagnosed with osteopetrosis in childhood and exhibited sclerotic changes, which were likely caused by osteopetrosis. Bone scintigraphy using Tc-99m-HMDP revealed no increase in tracer uptake in the jawbone. Finally, the patient was diagnosed with osteopetrosis-related left maxillary osteomyelitis and sinusitis; hence, she underwent hyperbaric oxygen therapy, multiple surgeries, and antibiotic therapy. Hepatic dysfunction occurred during the treatment, and a mediastinal tumor was suspected. Thus, fluorodeoxyglucosepositron emission tomography was performed. Further, the patient was diagnosed with extramedullary hematopoietic foci associated with osteopetrosis and hepatic dysfunction due to hepatitis B, there was no Tc-99m accumulation in the maxilla, and hepatitis was treated concurrently. Six months after the final surgery for osteomyelitis, antibiotic therapy was discontinued. Notably, 2 years have passed without exacerbation of osteomyelitis. Moreover, bone scintigraphy using Tc-99m-HMDP did not reveal increased Tc-99m accumulation in the jawbone. In cases of osteopetrosis-related osteomyelitis, interpretation of findings should be done cautiously because Tc-99m may accumulate in an area with active bone metabolism and osteomyelitis may be masked. Refractory osteomyelitis with osteopetrosis requires careful interpretation of MRI and bone scintigraphy scans, and long-term follow-up is required.