Primary intraosseous mucoepidermoid carcinoma (PIOC) is an infrequent malignancy of the head and neck that exhibits diverse biological behavior. The rareness of the location for a salivary gland tumor and the clinical and radiographic manifestations, suggestive of an odontogenic lesion often pose a diagnostic challenge. We hereby report such a unique case of central mucoepidermoid carcinoma that presented in an ambiguous manner, developing in the maxilla and intruding into the adjacent vital structures, adding a literature review.
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