. 2018 Mar 1;75(3):353-359.

doi: 10.1001/jamaneurol.2017.4317.

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

Daniel Martinez-Ramirez¹, Joohi Jimenez-Shahed², James Frederick Leckman³, Mauro Porta⁴, Domenico Servello⁵, Fan-Gang Meng⁶, Jens Kuhn^{7

8}, Daniel Huys⁷, Juan Carlos Baldermann⁷, Thomas Foltynie⁹, Marwan I Hariz^{9

10}, Eileen M Joyce⁹, Ludvic Zrinzo⁹, Zinovia Kefalopoulou⁹, Peter Silburn¹¹, Terry Coyne¹¹, Alon Y Mogilner¹², Michael H Pourfar¹², Suketu M Khandhar¹³, Man Auyeung¹⁴, Jill Louise Ostrem¹⁵, Veerle Visser-Vandewalle¹⁶, Marie-Laure Welter¹⁷, Luc Mallet^{18

19

20}, Carine Karachi²¹, Jean Luc Houeto^{22

23}, Bryan Timothy Klassen²⁴, Linda Ackermans²⁵, Takanobu Kaido^{26

27}, Yasin Temel²⁸, Robert E Gross²⁹, Harrison C Walker^{30

31}, Andres M Lozano³², Benjamin L Walter^{33

34

35}, Zoltan Mari³⁶, William S Anderson³⁶, Barbara Kelly Changizi³⁷, Elena Moro^{38

39}, Sarah Elizabeth Zauber⁴⁰, Lauren E Schrock⁴¹, Jian-Guo Zhang⁴², Wei Hu¹, Kyle Rizer¹, Erin H Monari¹, Kelly D Foote^{43

44}, Irene A Malaty¹, Wissam Deeb¹, Aysegul Gunduz⁴⁵, Michael S Okun^{1

44}

Affiliations

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

PMID: 29340590
PMCID: PMC5885852
DOI: 10.1001/jamaneurol.2017.4317

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

Daniel Martinez-Ramirez et al. JAMA Neurol. 2018.

. 2018 Mar 1;75(3):353-359.

doi: 10.1001/jamaneurol.2017.4317.

Authors

Affiliations

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

PMID: 29340590
PMCID: PMC5885852
DOI: 10.1001/jamaneurol.2017.4317

Erratum in

Erratum: Errors in Author Affiliations and Missing Degree.
[No authors listed] [No authors listed] JAMA Neurol. 2018 Mar;75(3):384. doi: 10.1001/jamaneurol.2018.0308. Epub 2018 Mar 12. JAMA Neurol. 2018. PMID: 31329657 Free PMC article.

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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Conflict of interest statement

Conflict of Interest Disclosures: Dr Leckman reported serving as an advisor for the European Multicentre Tics in Children Studies (EMTICS) and Tasly Pharmaceuticals LLC; receiving support from the National Institutes of Health (for research projects), the Tourette Association of America, and Grifols LLC; and receiving royalties for books published by John Wiley and Sons, McGraw-Hill, and Oxford University Press. Dr Okun reported serving as a consultant for the National Parkinson Foundation; receiving research grants from the National Institutes of Health, the National Parkinson Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the University of Florida Foundation; receiving grants R01 NR014852 and R01NS096008 from the National Institutes of Health for DBS research; receiving royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, and Cambridge (movement disorders books); serving as an associate editor for New England Journal of Medicine Journal Watch Neurology; participating in continuing medical education and educational activities on movement disorders (in the last 36 months) sponsored by PeerView, Prime, QuantiaMD, WebMD, Medicus, MedNet, Henry Stewart, and Vanderbilt University; and participating as a site principal investigator and/or coinvestigator for several National Institutes of Health, foundation, and industry-sponsored trials over the years but has not received honoraria. The institution and not Dr Okun received grants from Medtronic, Abbvie, Allergan, and Abbott/St Jude, and Dr Okun has no financial interest in these grants. No other disclosures were reported.

Figures

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References

1. Temel Y, Visser-Vandewalle V. Surgery in Tourette syndrome. Mov Disord. 2004;19(1):3-14. - PubMed
1. Vandewalle V, van der Linden C, Groenewegen HJ, Caemaert J. Stereotactic treatment of Gilles de la Tourette syndrome by high frequency stimulation of thalamus. Lancet. 1999;353(9154):724. - PubMed
1. Okun MS. Deep-brain stimulation for Parkinson’s disease. N Engl J Med. 2012;367(16):1529-1538. - PubMed
1. Miocinovic S, Somayajula S, Chitnis S, Vitek JL. History, applications, and mechanisms of deep brain stimulation. JAMA Neurol. 2013;70(2):163-171. - PubMed
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Efficacy and Safety of Deep Brain Stimulation in Tourette Syndrome: The International Tourette Syndrome Deep Brain Stimulation Public Database and Registry

Affiliations

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

Authors

Affiliations

Erratum in

Abstract

Conflict of interest statement

Figures

References

Publication types

MeSH terms

Grants and funding

LinkOut - more resources

Full Text Sources

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Medical