Efficacy and safety of deep brain stimulation in tourette syndrome the international tourette syndrome deep brain stimulation public database and registry

Daniel Martinez-Ramirez, Joohi Jimenez-Shahed, James Frederick Leckman, Mauro Porta, Domenico Servello, Fan Gang Meng, Jens Kuhn, Daniel Huys, Juan Carlos Baldermann, Thomas Foltynie, Marwan I. Hariz, Eileen M. Joyce, Ludvic Zrinzo, Zinovia Kefalopoulou, Peter Silburn, Terry Coyne, Alon Y. Mogilner, Michael H. Pourfar, Suketu M. Khandhar, Man Auyeung & 29 others Jill Louise Ostrem, Veerle Visser-Vandewalle, Marie Laure Welter, Luc Mallet, Carine Karachi, Jean Luc Houeto, Bryan Timothy Klassen, Linda Ackermans, Takanobu Kaido, Yasin Temel, Robert E. Gross, Harrison C. Walker, Andres M. Lozano, Benjamin L. Walter, Zoltan Mari, William S Anderson, Barbara Kelly Changizi, Elena Moro, Sarah Elizabeth Zauber, Lauren E. Schrock, Jian Guo Zhang, Wei Hu, Kyle Rizer, Erin H. Monari, Kelly D. Foote, Irene A. Malaty, Wissam Deeb, Aysegul Gunduz, Michael S. Okun

Research output: Contribution to journalArticle

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%]). MAINOUTCOMESANDMEASURES ScoresontheYale GlobalTicSeverity Scaleandadverseevents. 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 stimulationwas 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.

Original languageEnglish (US)
Pages (from-to)353-359
Number of pages7
JournalJAMA Neurology
Volume75
Issue number3
DOIs
StatePublished - Mar 1 2018

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Tourette Syndrome
Deep Brain Stimulation
Registries
Databases
Safety
Tics
Globus Pallidus
Internal Capsule
Self-Injurious Behavior
Dysarthria
Paresthesia
Intracranial Hemorrhages
Obsessive-Compulsive Disorder
Extremities

ASJC Scopus subject areas

  • Clinical Neurology

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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. / 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.

In: JAMA Neurology, Vol. 75, No. 3, 01.03.2018, p. 353-359.

Research output: Contribution to journalArticle

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 & Okun, MS 2018, 'Efficacy and safety of deep brain stimulation in tourette syndrome the international tourette syndrome deep brain stimulation public database and registry', JAMA Neurology, vol. 75, no. 3, pp. 353-359. https://doi.org/10.1001/jamaneurol.2017.4317
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. / Efficacy and safety of deep brain stimulation in tourette syndrome the international tourette syndrome deep brain stimulation public database and registry. In: JAMA Neurology. 2018 ; Vol. 75, No. 3. pp. 353-359.
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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{\%}]). MAINOUTCOMESANDMEASURES ScoresontheYale GlobalTicSeverity Scaleandadverseevents. 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 stimulationwas 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.",
author = "Daniel Martinez-Ramirez and Joohi Jimenez-Shahed and Leckman, {James Frederick} and Mauro Porta and Domenico Servello and Meng, {Fan Gang} and Jens Kuhn and Daniel Huys and Baldermann, {Juan Carlos} and Thomas Foltynie and Hariz, {Marwan I.} and Joyce, {Eileen M.} and Ludvic Zrinzo and Zinovia Kefalopoulou and Peter Silburn and Terry Coyne and Mogilner, {Alon Y.} and Pourfar, {Michael H.} and Khandhar, {Suketu M.} and Man Auyeung and Ostrem, {Jill Louise} and Veerle Visser-Vandewalle and Welter, {Marie Laure} and Luc Mallet and Carine Karachi and Houeto, {Jean Luc} and Klassen, {Bryan Timothy} and Linda Ackermans and Takanobu Kaido and Yasin Temel and Gross, {Robert E.} and Walker, {Harrison C.} and Lozano, {Andres M.} and Walter, {Benjamin L.} and Zoltan Mari and Anderson, {William S} and Changizi, {Barbara Kelly} and Elena Moro and Zauber, {Sarah Elizabeth} and Schrock, {Lauren E.} and Zhang, {Jian Guo} and Wei Hu and Kyle Rizer and Monari, {Erin H.} and Foote, {Kelly D.} and Malaty, {Irene A.} and Wissam Deeb and Aysegul Gunduz and Okun, {Michael S.}",
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T1 - Efficacy and safety of deep brain stimulation in tourette syndrome the international tourette syndrome deep brain stimulation public database and registry

AU - Martinez-Ramirez, Daniel

AU - Jimenez-Shahed, Joohi

AU - Leckman, James Frederick

AU - Porta, Mauro

AU - Servello, Domenico

AU - Meng, Fan Gang

AU - Kuhn, Jens

AU - Huys, Daniel

AU - Baldermann, Juan Carlos

AU - Foltynie, Thomas

AU - Hariz, Marwan I.

AU - Joyce, Eileen M.

AU - Zrinzo, Ludvic

AU - Kefalopoulou, Zinovia

AU - Silburn, Peter

AU - Coyne, Terry

AU - Mogilner, Alon Y.

AU - Pourfar, Michael H.

AU - Khandhar, Suketu M.

AU - Auyeung, Man

AU - Ostrem, Jill Louise

AU - Visser-Vandewalle, Veerle

AU - Welter, Marie Laure

AU - Mallet, Luc

AU - Karachi, Carine

AU - Houeto, Jean Luc

AU - Klassen, Bryan Timothy

AU - Ackermans, Linda

AU - Kaido, Takanobu

AU - Temel, Yasin

AU - Gross, Robert E.

AU - Walker, Harrison C.

AU - Lozano, Andres M.

AU - Walter, Benjamin L.

AU - Mari, Zoltan

AU - Anderson, William S

AU - Changizi, Barbara Kelly

AU - Moro, Elena

AU - Zauber, Sarah Elizabeth

AU - Schrock, Lauren E.

AU - Zhang, Jian Guo

AU - Hu, Wei

AU - Rizer, Kyle

AU - Monari, Erin H.

AU - Foote, Kelly D.

AU - Malaty, Irene A.

AU - Deeb, Wissam

AU - Gunduz, Aysegul

AU - Okun, Michael S.

PY - 2018/3/1

Y1 - 2018/3/1

N2 - 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%]). MAINOUTCOMESANDMEASURES ScoresontheYale GlobalTicSeverity Scaleandadverseevents. 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 stimulationwas 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.

AB - 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%]). MAINOUTCOMESANDMEASURES ScoresontheYale GlobalTicSeverity Scaleandadverseevents. 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 stimulationwas 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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