Case ReportsTardive Myoclonic Dyskinesia Responsive to Sodium OxybateD'Arrigo, Andrea Maria MD*†; Campiglio, Laura MD*; Casellato, Chiara MD*; Gambini, Chiara MD*; Belvedere, Daniela MD*; Secchi, Manuela MD*; Rosci, Chiara MD*; Priori, Alberto MD, PhD*†Author Information *Clinical Neurology Unit I, Department of Medicine, San Paolo University Hospital ASST Santi Paolo e Carlo; and †“Aldo Ravelli” Research Centre of Neurotechnology and Experimental Brain Therapeutics, Department of Health Sciences, University of Milan, Milan, Italy. Address correspondence and reprint requests to Alberto Priori, MD, PhD, Department of Health Sciences, Ospedale San Paolo, Via Antonio di Rudinì 8, Milan 20142, Italy; E-mail: email@example.com Conflicts of Interest and Source of Funding: The authors have no conflicts of interest and no sources of funding to declare. Clinical Neuropharmacology: 9/10 2018 - Volume 41 - Issue 5 - p 194-196 doi: 10.1097/WNF.0000000000000290 Buy Metrics Abstract 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. Copyright © 2018 Wolters Kluwer Health, Inc. All rights reserved.