Hyperkinetic movement disorders may be difficult to treat, but cases where patients respond to alcohol and/or drugs with similar effects have been described. We report the case of a 64-year-old man with tardive dyskinesia characterized by severe uncontrolled dystonic and myoclonic jerks of the face, shoulders, and arm and forearm muscles, which improved with oral sodium oxybate. Our case suggests the possibility to test sodium oxybate in patients with severe, drug-resistant hyperkinetic syndromes, especially when they are known to improve with alcohol.