Calcifying odontogenic cysts (COCs) are uncommon benign lesions arising from the odontogenic epithelium which are currently considered cysts. Histopathologically, it presents as an epithelial lining with ameloblastic characteristics, in addition to ghost cells with tendency to undergo calcifications. The objective of this study was to describe a case of a calcifying odontogenic cyst associated with odontoma, which was treated with a conservative two-step approach. A 29-year-old female presented with increased volume on the right side of the face with a lesion detected at the bottom of the buccal groove, extending from tooth 53 to 16. Radiographically, a wide, unilocular lesion was observed, involving tooth 13 that was impacted. Histopathological examination was consistent with COC associated with complex odontoma. No recurrence was seen at 6-month follow-up. The case addresses the importance of anatomopathological diagnosis of the lesion, since characterization of the cyst is fundamental to treatment
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