We report two unrelated patients affected of kinesigenic paroxysmal choreoathetosis (CPC), during a symptomatic period. Routine complementary exams were normal. The 99mTc-HMPAO cerebral SPECT showed hyperactivation in the basal ganglia opposite to the choreoathetosic symptoms and reduced untake in the near parietal and subcortical zones in one case. These observations suggest that abnormal hyperactivity of contralateral basal ganglia may cause choreathetotic movements in patients with CPC.