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 Volume 67, Number 6, September 26, 2006
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NEUROLOGY 2006;67:940-943
© 2006 American Academy of Neurology
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Severe tongue protrusion dystonia

Clinical syndromes and possible treatment

S. A. Schneider, MD, A. Aggarwal, DNB, M. Bhatt, DM, E. Dupont, MD, S. Tisch, MD, P. Limousin, MD, P. Lee, DM, N. Quinn, MD and K. P. Bhatia, MD

From the Sobell Department of Motor Neuroscience and Movement Disorders, Institute of Neurology, UCL, London, UK (S.A.S., N.Q., K.P.B.); Department of Movement Disorders, Jaslok Hospital and Research Center, Mumbai, India (A.A., M.B.); Department of Neurology, Aarhus University Hospital, Aarhus, Denmark (E.D.); Functional Neurosurgery Unit, Institute of Neurology, University College London, London, United Kingdom (S.T., P. Limousin); and Charles Dent Metabolic Unit, National Hospital for Neurology and Neurosurgery, Queen Square, London, UK (P. Lee).

Address correspondence and reprint requests to Dr. Kailash P. Bhatia, Sobell Department of Motor Neuroscience and Movement Disorders, Institute of Neurology, Queen Square, London WC1N 3BG, UK; e-mail: kbhatia{at}ion.ucl.ac.uk

We describe intermittent or sustained severe involuntary tongue protrusion in patients with a dystonic syndrome. Speech, swallowing, and breathing difficulties can be severe enough to be life threatening. Causes include neuroacanthocytosis, pantothenate kinase–associated neurodegeneration, Lesch–Nyhan syndrome, and postanoxic and tardive dystonia. The pathophysiology of intermittent severe tongue protrusion remains unknown. Tongue protrusion dystonia is often unresponsive to oral drugs but may benefit from botulinum toxin injections into the genioglossus muscle. Bilateral deep brain pallidal stimulation was beneficial in two cases.

Additional material related to this article can be found on the Neurology Web site. Go to www.neurology.org and scroll down the Table of Contents for the September 26 issue to find the title link for this article.

S.A.S. was supported by the Brain Research Trust, United Kingdom.

Disclosure: The authors report no conflicts of interest.

Received February 21, 2006. Accepted in final form May 9, 2006.






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