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

Author

Martinez-Ramirez, Daniel, Jimenez-Shahed, Joohi, Leckman, James Frederick, Porta, Mauro, Servello, Domenico, Meng, Fan-Gang, Kuhn, Jens, Huys, Daniel, Baldermann, Juan Carlos, Foltynie, Thomas, Hariz, Marwan I., Joyce, Eileen M., Zrinzo, Ludvic, Kefalopoulou, Zinovia, Silburn, Peter, Coyne, Terry, Mogilner, Alon Y., Pourfar, Michael H., Khandhar, Suketu M., Auyeung, Man, Ostrem, Jill Louise, Visser-Vandewalle, Veerle, Welter, Marie-Laure, Mallet, Luc, Karachi, Carine, Houeto, Jean Luc, Klassen, Bryan Timothy, Ackermans, Linda, Kaido, Takanobu, Temel, Yasin, Gross, Robert E., Walker, Harrison C., Lozano, Andres M., Walter, Benjamin L., Mari, Zoltan, Anderson, William S., Changizi, Barbara Kelly, Moro, Elena, Zauber, Sarah Elizabeth, Schrock, Lauren E., Zhang, Jian-Guo, Hu, Wei, Rizer, Kyle, Monari, Erin H., Foote, Kelly D., Malaty, Irene A., Deeb, Wissam, Gunduz, Aysegul, Okun, Michael S., Martinez-Ramirez, Daniel, Jimenez-Shahed, Joohi, Leckman, James Frederick, Porta, Mauro, Servello, Domenico, Meng, Fan-Gang, Kuhn, Jens, Huys, Daniel, Baldermann, Juan Carlos, Foltynie, Thomas, Hariz, Marwan I., Joyce, Eileen M., Zrinzo, Ludvic, Kefalopoulou, Zinovia, Silburn, Peter, Coyne, Terry, Mogilner, Alon Y., Pourfar, Michael H., Khandhar, Suketu M., Auyeung, Man, Ostrem, Jill Louise, Visser-Vandewalle, Veerle, Welter, Marie-Laure, Mallet, Luc, Karachi, Carine, Houeto, Jean Luc, Klassen, Bryan Timothy, Ackermans, Linda, Kaido, Takanobu, Temel, Yasin, Gross, Robert E., Walker, Harrison C., Lozano, Andres M., Walter, Benjamin L., Mari, Zoltan, Anderson, William S., Changizi, Barbara Kelly, Moro, Elena, Zauber, Sarah Elizabeth, Schrock, Lauren E., Zhang, Jian-Guo, Hu, Wei, Rizer, Kyle, Monari, Erin H., Foote, Kelly D., Malaty, Irene A., Deeb, Wissam, Gunduz, Aysegul, and Okun, Michael S.

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%, En rättelse har publicerats: / A correction has been published:Error in abstract. (2018). JAMA Neurology, 75(3), 384-384. doi:10.1001/jamaneurol.2018.0060

Published
2018
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2. Efficacy and Safety of Deep Brain Stimulation in Tourette Syndrome: The International Tourette Syndrome Deep Brain Stimulation Public Database and Registry.

Author

Martinez-Ramirez D, Jimenez-Shahed J, Leckman JF, Porta M, Servello D, Meng FG, Kuhn J, Huys D, Baldermann JC, Foltynie T, Hariz MI, Joyce EM, Zrinzo L, Kefalopoulou Z, Silburn P, Coyne T, Mogilner AY, Pourfar MH, Khandhar SM, Auyeung M, Ostrem JL, Visser-Vandewalle V, Welter ML, Mallet L, Karachi C, Houeto JL, Klassen BT, Ackermans L, Kaido T, Temel Y, Gross RE, Walker HC, Lozano AM, Walter BL, Mari Z, Anderson WS, Changizi BK, Moro E, Zauber SE, Schrock LE, Zhang JG, Hu W, Rizer K, Monari EH, Foote KD, Malaty IA, Deeb W, Gunduz A, and Okun MS

Subjects
Adolescent, Adult, Cohort Studies, Databases, Factual statistics & numerical data, Female, Globus Pallidus physiology, Humans, International Cooperation, Male, Middle Aged, Severity of Illness Index, Single-Blind Method, Thalamus physiology, Young Adult, Deep Brain Stimulation methods, Registries, Tourette Syndrome therapy, Treatment Outcome
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.

Published
2018
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