Adrenocortical carcinoma (ACC) is an uncommon primary cancer in the adrenal gland. Its incidence of showing metastasis in the head and neck region is very rare. Herein, we present a case of a 46-year-old man who presented with complaints of pain and numbness on the left side of the lower face for 4 months. Radiographic examination revealed an osteolytic lesion with an ill-defined border in the left body region of the mandible. Histopathologic examination revealed a tumor composed of sheets of oval to polygon-shaped tumor cells predominantly displaying abundant eosinophilic granular cytoplasm. These tumor cells showed features of a high degree of anaplasia. On immunohistochemical examination, tumor cells were focally positive for synaptophysin, inhibin, vimentin, pancytokeratin (pan-CK), cytokeratin (CK)5/6, CD68, and CK8/18 and immunonegative for CK7, chromogranin, melan-A, S100, SMA, and SATB2. The Ki-67 proliferation index was approximately 20%. To the best of our knowledge, this is the first case of metastatic oncocytic ACC to the oral cavity region.
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