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A case of a xanthogranuloma arising in the anterior skull base involving the bone of the left orbital roof, the dura mater, and the periorbit is presented. The lesion was completely removed by a supraorbital “eyebrow” skin incision using frameless stereotactic image guidance. A reconstruction of the orbital roof was performed using a titanium mesh graft. The neuropathological investigation revealed all the characteristics of a xanthogranuloma. Intracranially, xanthogranulomas appear as rare tumors of the dura or choroid plexus, usually arising in association with histocytosis X or familial hyperlipoproteinemia. One case not associated with these diseases is discussed.