Hypertensive encephalopathy and basal ganglia intracerebral hemorrhage (ICH) are a medical emergency caused by a sudden elevation of systemic blood pressure. Although the relationship between hypertensive encephalopathy and large ICH has not been clarified yet, Cushing reflex in acute elevations of ICP due to large ICH may induce or aggravate hypertensive encephalopathy. We report a rare case of isolated hypertensive brainstem encephalopathy combined with hypertensive ICH.