Osteolipoma is an ossified lipoma with distinct components of fat and bone. We present a case of interhemispheric osteolipoma associated with total agenesis of the corpus callosum. A 20-year-old man complained of severe headache, nausea and vomiting. Brain computed tomography showed a low-density mass in an interhemispheric fissure, with high T1 and T2 magnetic resonance signals compatible with fat. The mass measured 4.9 x 2.9 cm in size and showed peripheral calcifications. There was another small piece of same signal mass within the lateral ventricular choroid plexus. The interhemispheric lesion was removed by an interhemispheric approach. Osteolipoma is rare in interhemispheric region, however, it should be a differential diagnosis of lesions with fat intensity mass and calcifications.