Ossifying fibroma is a benign fibro-osseous tumor originating from undifferentiated periodontal ligament cells. This report aims to present a case of infected ossifying fibroma mimicking chronic osteomyelitis. A sixty-year-old female presented with a history of continuously growing mass on her right jaw from four years ago with purulent discharge since last year. The extraoral examination revealed an oro-cutaneous fistula with ongoing purulent discharge in the right submandibular region. The intraoral examination revealed exposed bone with pus on the right mandibular edentulous ridge. The panoramic radiograph and MSCT imaging showed sequestrum and involucrum formation with the impression of chronic osteomyelitis on the right mandible. However, the subsequent histopathological examination showed a diagnosis of ossifying fibroma. Tumor excision and fistulectomy under general anesthesia were performed to treat this tumor. The post-operative histopathological examination showed the proliferation of fibroblasts accompanied by woven bone and osteoblastic rimming, confirming the diagnosis of ossifying fibroma. The evaluation and follow-up in the first, second, 6th, and 12th months after surgery demonstrated no recurrence and infection. Infected ossifying fibroma can mimic the clinical features of chronic osteomyelitis. The diagnosis of ossifying fibroma should be supported with histopathological data to determine the accurate management.
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