Osteolipoma is a benign neoplasm originating from the mesenchymal fat tissue cells and characterized by the presence of bone formation. In the head and neck district osteolipoma has been described as parapharyngeal, nasopharinx, submandibular gland, eyelid, floor of the mouth, and skull base lesion. Only 1 case of osteolipoma was reported as a neoplasm of the parotid region. Considering the rarity of the osteolipoma, our purpose was to describe the first case of totally intraparotid gland localization of osteolipoma and summarize and review the actual literature concerning the presence of this tumor in the region of major salivary glands. A 56-years-old male patient was treated for intraparotid osteolipoma. Surgical removal of tumor was done with a standard preauricular/cervical incision and blunt dissection. The facial nerve was identified and preserved completely. In the reviewed series, all cases were reported in adult patients with a relative long history of slow progression. According to our paper and the review of the literature, we can assess that Osteolipoma is an extremely rare lesion affecting the major salivary glands, which should be considered along with common salivary gland tumors in the differential diagnosis.
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