A 28 year-old man with chronic hepatitis B was administered interferon-alpha (5 x 10(6) IU) intramuscularly once a day for 28 days. Eight days after the end of interferon treatment, he showed signs of inability to sit still for ten seconds and walked around constantly. His akathisia symptoms worsened thereafter. Clonazepam, thioridazine, beta-blockers, anticholinergics, and bromocriptine did not ameliorate his akathisia. Two days' administration of levomepromazine 100 mg led him to a coma for 2 days. Intravenous levodopa 25 mg ameliorated his akathisia symptoms drastically. He became completely premorbid 2 weeks after administration of levodopa. The present report illustrates a rare case of refractory akathisia after interferon-alpha treatment and also that levodopa treatment would be theoretically and practically useful in reducing the neurotoxicity associated with interferon-alpha.