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Original Investigation
March 2018

Efficacy and Safety of Deep Brain Stimulation in Tourette SyndromeThe International Tourette Syndrome Deep Brain Stimulation Public Database and Registry

Author Affiliations
  • 1Center for Movement Disorders and Neurorestoration, Department of Neurology, University of Florida, Gainesville
  • 2Parkinson’s Disease Center and Movement Disorders Clinic, Baylor College of Medicine, Houston, Texas
  • 3Yale Child Study Center, Yale University School of Medicine, New Haven, Connecticut
  • 4Tourette’s Syndrome and Movement Disorders Center, Galeazzi Hospital, Milan, Italy
  • 5Neurosurgical Department, Isituto Ortopedico Galeazzi, Milan, Italy
  • 6Department of Functional Neurosurgery, Beijing Neurosurgical Institute, Capital Medical University, Beijing, China
  • 7Department of Psychiatry and Psychotherapy, University of Cologne, Cologne, Germany
  • 8Department of Psychiatry, Psychotherapy, and Psychosomatic Medicine, Johanniter Hospital Oberhausen, Oberhausen, Germany
  • 9Sobell Department of Motor Neuroscience, University College London Institute of Neurology, London, United Kingdom
  • 10Department of Clinical Neuroscience, Umeå University, Umeå, Sweden
  • 11Queensland Brain Institute, University of Queensland, Brisbane, Queensland, Australia
  • 12Department of Neurosurgery, Center for Neuromodulation, New York University Langone Medical Center, New York
  • 13Department of Neurology, The Permanente Medical Group (Kaiser Permanente Northern California), Comprehensive Movement Disorders Program, Sacramento, California
  • 14Department of Medicine, Pamela Youde Nethersole Eastern Hospital, Hong Kong, SAR China
  • 15Department of Neurology, University of California, San Francisco
  • 16Department of Stereotaxy and Functional Neurosurgery, University Hospital Cologne, Cologne, Germany
  • 17The French National Institute of Health and Medical Research U 1127, The National Center for Scientific Research 7225, Sorbonne Universités, University of Pierre and Marie Curie University of Paris 06 UMR S 1127, Institut du Cerveau et de la Moëlle Epinière, The Brain and Spinal Cord Institute, Paris, France
  • 18Assistance Publique–Hôpitaux de Paris, Personalised Neurology and Psychiatry University Department, Hôpitaux Universitaires Henri Mondor–Albert Chenevier, Université Paris–Est Créteil, Créteil, France
  • 19Sorbonne Universités, University of Pierre and Marie Curie University of Paris 06, CNRS, INSERM, Institut du Cerveau et de la Moëlle épinière, Paris, France
  • 20Department of Mental Health and Psychiatry, Global Health Institute, University of Geneva, Geneva, Switzerland
  • 21Institut du Cerveau et de la Moëlle Epinière, The French National Institute of Health and Medical Research U 1127, The National Center for Scientific Research 7225, Sorbonne Universités, University of Paris 06, UMR S 1127 Paris, France; Department of Neurosurgery, Pitié-Salpêtrière Hospital, Paris, France
  • 22Service de Neurologie, Centers for Clinical Investigation 1402, Centre Hospitalier Universitaire de Poitiers, Poitiers, France
  • 23Université de Poitiers, Poitiers, France
  • 24Department of Neurology, Mayo Clinic, Rochester, Minnesota
  • 25Department of Neurosurgery, Maastricht University Medical Centre, Maastricht, the Netherlands
  • 26Department of Neurosurgery, National Hospital Organization Nara Medical Center, Nara, Japan
  • 27Anatomy and Physiology Laboratory, Department of Health and Nutrition, Osaka Shoin Women’s University, Osaka, Japan
  • 28Maastricht University Medical Center, Maastricht, the Netherlands; MHeNs, Experimental Neurosurgery, Maastricht University, Maastricht, the Netherlands
  • 29Emory University, Atlanta, Georgia
  • 30Division of Movement Disorders, Department of Neurology, University of Alabama at Birmingham
  • 31Department of Biomedical Engineering, University of Alabama at Birmingham
  • 32Toronto Western Hospital, Division of Neurosurgery, University of Toronto, Toronto, Ontario, Canada
  • 33School of Medicine, Case Western Reserve University, Cleveland, Ohio
  • 34US Department of Veterans Affairs, Louis Stokes Cleveland Veterans Affairs Medical Center, Functional Electrical Stimulation Center of Excellence, Rehabilitation R&D Service, Cleveland, Ohio
  • 35Department of Neurology, University Hospitals Cleveland Medical Center, Cleveland, Ohio
  • 36Department of Neurology and Neurosurgery, Johns Hopkins University, Baltimore, Maryland
  • 37Department of Neurology, The Ohio State University Wexner Medical Center, Columbus
  • 38Division of Neurology, Centre Hospitalier Universitaire de Grenoble, Grenoble, France
  • 39Grenoble Alpes University, Grenoble, France
  • 40Department of Neurology, Indiana University School of Medicine, Indianapolis
  • 41Neuromodulation Research Center, Department of Neurology, University of Minnesota, Minneapolis
  • 42Department of Functional Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
  • 43Center for Movement Disorders and Neurorestoration, Department of Neurosurgery, University of Florida, Gainesville
  • 44Fixel Center for Neurological Diseases, Gainesville, Florida
  • 45Brain Map Lab, Center for Movement Disorders and Neurorestoration, Department of Neurology, University of Florida, Gainesville
JAMA Neurol. 2018;75(3):353-359. doi:10.1001/jamaneurol.2017.4317
Key Points

Question  What are the outcomes associated with deep brain stimulation in Tourette syndrome?

Findings  In this study including 185 patients from 10 countries in the International Deep Brain Stimulation Database and Registry, the mean Yale Global Tic Severity Scale score improved 45.1% at 1 year after deep brain stimulation implantation. The centromedian thalamic region was the most common target of deep brain stimulation implantation (57.1%), with no differences observed between targets, while the most common adverse events were dysarthria (6.3%) and paresthesias (8.2%), with hemorrhages occurring in 1.3% of patients, infection in 3.2%, and explantation in 0.6%.

Meaning  Deep brain stimulation was safe and associated with clinical improvements; a publicly available website has been released that tracks the outcomes among all participating centers.

Abstract

Importance  Collective evidence has strongly suggested that deep brain stimulation (DBS) is a promising therapy for Tourette syndrome.

Objective  To assess the efficacy and safety of DBS in a multinational cohort of patients with Tourette syndrome.

Design, Setting, and Participants  The prospective International Deep Brain Stimulation Database and Registry included 185 patients with medically refractory Tourette syndrome who underwent DBS implantation from January 1, 2012, to December 31, 2016, at 31 institutions in 10 countries worldwide.

Exposures  Patients with medically refractory symptoms received DBS implantation in the centromedian thalamic region (93 of 163 [57.1%]), the anterior globus pallidus internus (41 of 163 [25.2%]), the posterior globus pallidus internus (25 of 163 [15.3%]), and the anterior limb of the internal capsule (4 of 163 [2.5%]).

Main Outcomes and Measures  Scores on the Yale Global Tic Severity Scale and adverse events.

Results  The International Deep Brain Stimulation Database and Registry enrolled 185 patients (of 171 with available data, 37 females and 134 males; mean [SD] age at surgery, 29.1 [10.8] years [range, 13-58 years]). Symptoms of obsessive-compulsive disorder were present in 97 of 151 patients (64.2%) and 32 of 148 (21.6%) had a history of self-injurious behavior. The mean (SD) total Yale Global Tic Severity Scale score improved from 75.01 (18.36) at baseline to 41.19 (20.00) at 1 year after DBS implantation (P < .001). The mean (SD) motor tic subscore improved from 21.00 (3.72) at baseline to 12.91 (5.78) after 1 year (P < .001), and the mean (SD) phonic tic subscore improved from 16.82 (6.56) at baseline to 9.63 (6.99) at 1 year (P < .001). The overall adverse event rate was 35.4% (56 of 158 patients), with intracranial hemorrhage occurring in 2 patients (1.3%), infection in 4 patients with 5 events (3.2%), and lead explantation in 1 patient (0.6%). The most common stimulation-induced adverse effects were dysarthria (10 [6.3%]) and paresthesia (13 [8.2%]).

Conclusions and Relevance  Deep brain stimulation was associated with symptomatic improvement in patients with Tourette syndrome but also with important adverse events. A publicly available website on outcomes of DBS in patients with Tourette syndrome has been provided.

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