Peripheral ameloblastoma (PA) refers to a rare neoplasm that accounts for approximately 1%–5% of all ameloblastomas. It arises in the soft tissues overlying a tooth-bearing region, and those found in nontooth-bearing locations, such as the buccal mucosa, lips, and palate. This article aims at describing a case of a rare form of extragingival PA in the nontooth-bearing region and challenges in its management in settings with limited resources. A 55-year-old African male presented with a painless swelling on the right cheek for about 6 years, which was clinically diagnosed as pleomorphic adenoma; however, both pre- and postoperative histopathological results reported the swelling to be follicular ameloblastoma. Extragingival PA is a rare variant of ameloblastoma, which needs to be included as one of the differential diagnoses of the buccal mucosal swellings.
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