Treatment of tardive dystonia with oral baclofen produces ambivalent and overall disappointing results. However, because only a small proportion of the baclofen penetrates into the central nervous system when administered orally, we tested whether it is possible to increase the efficacy of treatment by continuous intrathecal infusion of baclofen (CITB) in a patient with severe tardive axial dystonia unresponsive to conventional therapy. A dose of 100 micrograms/day improved muscle tone, head control, posture, and walking distance; electromyography showed a marked decrease of dystonic muscle activity with fully preserved voluntary muscle activity, and the patient reported substantial reduction of pain. Apart from some minor discomfort at the site of operation, no side effects were noted. Further studies should be encouraged to evaluate the usefulness of CITB for other patients with severe and otherwise untreatable tardive dystonia.