We presented a case of severe postoperative choreoathetosis which occurred in a 3-year-old boy with tetralogy of Fallot early postoperatively but almost completely recovered within two years after the operation. Because of the large coronary arterial branch on the right ventricular outflow, a small outflow incision and deep hypothermia (lowest rectal temperature was 13 degrees C) and short duration of circulatory arrest (8 minutes) were adopted. Postoperative course was uneventful till the onset of choreoathetosis on the fifth postoperative day. His symptoms and signs of choreoathetosis, oral-facial dyskinesias, hypotonia, affective changes and also pseudobulbar signs were becoming serious during the first week from the onset, but afterwards his condition started getting better gradually every week, and every month. Now, 20 months after the operation, he is almost completely recovered except for small and slow involuntary movements. Though investiations including CT, MRI and EEG were all almost normal, regional nonspecific low area of the frontal lobe and cerebellum was detected by SPECT (single photon emission computed tomography) on the 32nd and 94th postoperative days, respectively.
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