A leiomyoma is a benign neoplasm composed of smooth muscle cells that commonly occurs in the genitourinary and gastrointestinal tracts. Primary intracranial leiomyoma, however, is extremely rare and only a few cases have been reported to date. In this study, we present two cases of primary intracranial leiomyomas in middle-aged men. Magnetic resonance images (MRI) showed lesions with homogeneous low signals on T1-, T2-, diffusion-weighted, and FLAIR sequences. Tumors were totally removed and there was no evidence of recurrence in the follow-up study. Pathological analysis with immunohistochemistry revealed that tumors had characteristics of benign smooth muscles. A review of relevant literature has been conducted.