BACKGROUND: Unlike delayed pain syndrome, acute central pain is a very rare symptom in acute stroke. In addition, the incidence of hemiballismus in acute cerebrovascular diseases is less than 1%. To our knowledge, the association of these 2 clinical conditions has not been previously described. PATIENT AND METHODS: After observing one patient with hemiballismus accompanied by ipsilateral acute limb pain at stroke onset, we retrospectively examined more than 4000 patients in the Lausanne Stroke Registry for hemiballismus-hemichorea occurring together with acute ipsilateral pain. RESULTS: Of the 29 subjects with hemiballismus-hemichorea, the observed patient was the only one to have acute pain at the onset of stroke. Magnetic resonance imaging showed acute infarction in the territory of the right anterior parietal artery, whereas the basal ganglia, thalamus, and subthalamic region were intact. CONCLUSIONS: The syndrome of acute limb pain associated with hemiballismus may result from disconnection of the parietal lobe from deeper structures. In contrast with isolated hemiballismus, we suggest that the simultaneous occurrence of this movement disorder with ipsilateral pain is specific for an anterior parietal artery stroke.