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Ameloblastomas are low-grade neoplasms of odontogenic epithelium that account for about 1% of all oral tumors and about 10% of odontogenic tumors. Rarely, these tumors have a potential of distance metastasis, and once they do metastasize, they are termed as metastatic ameloblastoma. This article aims at describing a case of a rare form of maxillary ameloblastoma that metastasized to the lung and challenges in its management in settings with limited resources. A 24-year-old African male presented with a massive swelling on the right side of the face for 2 years in 2018. He was previously operated on for a swelling on the right side of the maxilla in 2012, which was histologically diagnosed as ameloblastoma. Clinical and radiological evaluation revealed a massive maxillary tumor that had a local extension to the brain. X-ray and computed tomography (CT) scan of chest pointed out to metastatic mass in the lungs. Histology of the recurrent tumor mass revealed it to be follicular ameloblastoma, and CT-guided fine-needle aspiration cytology of the lung mass showed microscopic features of ameloblastoma similar to the primary jaw tumor.
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