A 25-year-old man had angiolipoma of the left thalamus producing sudden severe headache, coma, right hemiparalysis, and aphasia as usually observed in cerebrovascular disease. The patient remained right hemiparalytic and aphasic after removal of the thalamic lesion. A vascular lesion was also detected angiographically in the left lateral ventricular choroid plexus but was not excised. Only 3 intracranial angiolipomas including the current instance were on record. However, it was only in our patient that the lesion suddenly presented with intracerebral hemorrhage. The pathogenesis of the angiolipoma was suggested to be associated with maldifferentiation of mesenchyme and/or its derivatives toward the vascular and adipose tissue within the same mass, resulting in the formation of the angiolipoma.