Xanthomas are benign soft-tissue lesions that frequently occur on the skin after minor trauma or friction. Intraosseous xanthomas are infrequent and the associated literature is scarce; it mainly addresses cases in the facial mass. Imaging and clinical examinations do not yield a specific diagnosis, so histopathology and immunohistochemistry are used to confirm the diagnosis of primary intraosseous xanthoma. Here, we present the case of a 46-year-old male diagnosed with primary intraosseous xanthoma of the frontal bone, describing the clinical, imaging and histological development and his conservative surgical therapeutic management without recurrence after clinical and imaging controls.