We report a rapid, dramatic and sustained improvement following bilateral pallidal stimulation in two patients affected by intractable generalized tardive dystonia. Both patients had a chronic psychiatric disorder and developed chronic disabling generalized dystonic symptoms persisting despite prolonged withdrawal of neuroleptics and all available symptomatic treatment. The clinical benefit in both patients persisted throughout all the follow up period of 13 and 7 months. The favorable and prolonged response in our two patients suggests that deep brain stimulation may be an effective treatment for medically refractory tardive dystonia.