A 79-year-old woman with hypertension was evaluated 3 hours and 20 minutes after the sudden onset of left-sided weakness which lasted about 15 minutes and was followed by involuntary, coarse, flinging movements of the left extremities (hemiballistic), occurring every few minutes, and facial asymmetry. Brain computed tomography revealed no abnormalities. The patient received intravenous thrombolysis with 0.9 mg/kg of alteplase 4 hours after the symptom onset. Involuntary movements and central facial nerve paresis subsided within 48 hours of the thrombolysis. Magnetic resonance imaging at day 5 revealed restricted diffusion within the right globus pallidus, which was a new ischemic lesion. Thrombolysis should be considered in hemiballism as a presenting symptom of acute stroke.