We report a case of primary intracranial leiomyoma in 29-year-old woman presented with severe headache. The radiology diagnosis was consistent with meningioma. However, histologically, the tumor had the characteristic appearance of benign smooth muscle. This was confirmed by immunohistochemistry and electron microscopy. Benign metastasizing leiomyoma was excluded by thorough imaging. Although rare, leiomyoma should be considered in the differential diagnosis of well-circumscribed intracranial lesion.