Prospective randomized double-blind trial of bilateral thalamic deep brain stimulation in adults with Tourette syndrome

Robert J. Maciunas M.D., M.P.H.1, Brian N. Maddux M.D., Ph.D.2, David E. Riley M.D.2, Christina M. Whitney R.N.C.S., D.N.Sc.2, Mike R. Schoenberg Ph.D.2, Paula J. Ogrocki Ph.D.2, Jeffrey M. Albert Ph.D.3, and Deborah J. Gould M.D.4
View More View Less
  • 1 Departments of Neurosurgery and
  • | 2 Neurology, Neurological Institute, University Hospitals Case Medical Center;
  • | 3 Department of Epidemiology and Biostatistics, Case Western Reserve University, Cleveland; and
  • | 4 Northeast Ohio Health Services, Beachwood, Ohio
Restricted access

Purchase Now

USD  $45.00

JNS + Pediatrics - 1 year subscription bundle (Individuals Only)

USD  $505.00

JNS + Pediatrics + Spine - 1 year subscription bundle (Individuals Only)

USD  $600.00
Print or Print + Online

Object

The severity of Tourette syndrome (TS) typically peaks just before adolescence and diminishes afterward. In some patients, however, TS progresses into adulthood, and proves to be medically refractory. The authors conducted a prospective double-blind crossover trial of bilateral thalamic deep brain stimulation (DBS) in five adults with TS.

Methods

Bilateral thalamic electrodes were implanted. An independent programmer established optimal stimulator settings in a single session. Subjective and objective results were assessed in a double-blind randomized manner for 4 weeks, with each week spent in one of four states of unilateral or bilateral stimulation. Results were similarly assessed 3 months after unblinded bilateral stimulator activation while repeated open programming sessions were permitted.

Results

In the randomized phase of the trial, a statistically significant (p < 0.03, Friedman exact test) reduction in the modified Rush Video-Based Rating Scale score (primary outcome measure) was identified in the bilateral on state. Improvement was noted in motor and sonic tic counts as well as on the Yale Global Tic Severity Scale and TS Symptom List scores (secondary outcome measures). Benefit was persistent after 3 months of open stimulator programming. Quality of life indices were also improved. Three of five patients had marked improvement according to all primary and secondary outcome measures.

Conclusions

Bilateral thalamic DBS appears to reduce tic frequency and severity in some patients with TS who have exhausted other available means of treatment.

Abbreviations used in this paper:

BDI-2 = Beck Depression Inventory; DBS = deep brain stimulation; FDA = Food and Drug Administration; GPI = globus pallidus internus; HAM-A = Hamilton Rating Scale for Anxiety; HAM-D = Hamilton Rating Scale for Depression; MR = magnetic resonance; mRVRS = modified Rush Video-Based Rating Scale; SD = standard deviation; SF-36 = 36-Item Short Form Health Survey; TS = Tourette syndrome; TSSL = TS Symptom List; VAS = visual analog scale; Y-BOCS = Yale-Brown Obsessive Compulsive Scale; YGTSS = Yale Global Tic Severity Scale.

JNS + Pediatrics - 1 year subscription bundle (Individuals Only)

USD  $505.00

JNS + Pediatrics + Spine - 1 year subscription bundle (Individuals Only)

USD  $600.00

Contributor Notes

Address correspondence to: Robert J. Maciunas, M.D., M.P.H., Department of Neurosurgery, Neurological Institute, University Hospitals Case Medical Center, 11100 Euclid Avenue, Cleveland, Ohio 44106. email: robert.maciunas@uhhospitals.org.
  • 1

    Ackermans L, , Temel Y, , Cath D, , van der Linden C, , Bruggerman R, & Kleijer M, et al. : Deep brain stimulation in Tourette's syndrome: two targets?. Mov Disord 21:709713, 2006

    • Search Google Scholar
    • Export Citation
  • 2

    American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders ed 4 Washington, DC, American Psychiatric Association, 1994

    • Search Google Scholar
    • Export Citation
  • 3

    Beck AT, , Steer RA, & Brown GK: BDI-II. Manual for the Beck Depression Inventory ed 2 San Antonio, TX, The Psychological Corporation, 1996

  • 4

    Bloch MH, , Peterson BS, , Scahill L, , Otka J, , Katsovich L, & Zhang H, et al. : Adulthood outcome of tic and obsessive-compulsive symptom severity in children with Tourette syndrome. Arch Pediatr Adolesc Med 160:6569, 2006

    • Search Google Scholar
    • Export Citation
  • 5

    Channon S, , Pratt P, & Robertson MM: Executive function, memory, and learning in Tourette's syndrome. Neuropsychology 17:247254, 2003

  • 6

    Cooper IS: Dystonia reversal by operation on basal ganglia. Arch Neurol 7:132145, 1962

  • 7

    Cooper IS: Involuntary Movement Disorders New York, Hoeber Medical Division, 1969. 274279

  • 8

    Corkum PV, & Siegel LS: Is the continuous performance task a valuable research tool for hyperactivity disorder?. J Child Psychol Psychiatry 34:12171239, 1993

    • Search Google Scholar
    • Export Citation
  • 9

    Deuschl G, , Herzog J, , Kleiner-Fisman G, , Kubu C, , Lozano A, & Lyons KE, et al. : Deep brain stimulation: postoperative issues. Mov Disord 21:14 Suppl S219S237, 2006

    • Search Google Scholar
    • Export Citation
  • 10

    Diederich NJ, , Kalteis K, , Stamenkovic M, , Pieri V, & Alesch F: Efficient internal pallidal stimulation in Gilles de la Tourette syndrome: a case report. Mov Disord 20:14961499, 2005

    • Search Google Scholar
    • Export Citation
  • 11

    Flaherty AW, , Eskandar EN, , Williams ZM, , Cebula C, & Cosgrove GR: Novel control of tics, mood, and creativity with internal capsule and nucleus accumbens stimulation. Neurology 62:5 Suppl P02.141 2004. (Abstract)

    • Search Google Scholar
    • Export Citation
  • 12

    Georgiou N, , Bradshaw JL, , Phillips JG, , Bradshaw JA, & Chiu E: The Simon effect and attention deficits in Gilles de la Tourette's syndrome and Huntington's disease. Brain 118:13051318, 1995

    • Search Google Scholar
    • Export Citation
  • 13

    Goetz CG, & Kompoliti K: Rating scales and quantitative assessment of tics. Adv Neurol 85:3142, 2001

  • 14

    Goetz CG, , Pappert EJ, , Raman R, & Leurgans S: Advantages of a modified version of an objective video-based rating scale for Gilles de la Tourette syndrome. Mov Disord 14:502506, 1999

    • Search Google Scholar
    • Export Citation
  • 15

    Goetz CG, , Tanner CM, , Stebbins GT, , Leipzig G, & Carr WC: Adult tics in Gilles de la Tourette's syndrome: description and risk factors. Neurology 42:784788, 1992

    • Search Google Scholar
    • Export Citation
  • 16

    Goetz CG, , Tanner CM, , Wilson RS, & Shannon KM: A rating scale for Gilles de la Tourette's syndrome: description, reliability, and validity data. Neurology 37:15421544, 1987

    • Search Google Scholar
    • Export Citation
  • 17

    Goodman WK, , Price LH, , Rasmussen SA, , Mazure C, , Fleischmann RL, & Hill CL, et al. : The Yale-Brown Obsessive Compulsive Scale: I. Development, use, and reliability. Arch Gen Psychiatry 46:10061011, 1989

    • Search Google Scholar
    • Export Citation
  • 18

    Hamilton M: The assessment of anxiety states by rating. Br J Med Psychol 32:5255, 1959

  • 19

    Hamilton M: Development of a rating scale for primary depressive illness. Br J Soc Clin Psychol 6:278296, 1967

  • 20

    Hassler R, Stereotaxic surgery for psychiatric disturbances. Schaltenbrand G, & Walker AE: Stereotaxy of the Human Brain ed 2 New York, Thieme-Stratton, 1982. 570590

    • Search Google Scholar
    • Export Citation
  • 21

    Hassler R, & Dieckmann G, Relief of obsessive-compulsive disorders, phobias, and tics by stereotactic coagulations of the rostral intralaminar and medial-thalamic nuclei. Laitinen LV, & Livingston K: Surgical Approaches in Psychiatry. Proceedings of the 3rd International Congress of Psychosurgery Cambridge, UK Garden City Press, 1973. 206212

    • Search Google Scholar
    • Export Citation
  • 22

    Hathaway SR, & McKinley JC: Minnesota Multiphasic Personality Inventory—2nd Edition (MMPI-2): Manual for Administration and Scoring Minneapolis, University of Minnesota Press, 1989

    • Search Google Scholar
    • Export Citation
  • 23

    Hornse H, , Banerjee S, , Zeitlin H, & Robertson M: The prevalence of Tourette syndrome in 13-14–year-olds in mainstream schools. J Child Psychol Psychiatry 42:10351039, 2001

    • Search Google Scholar
    • Export Citation
  • 24

    Houeto JL, , Karachi C, , Mallet L, , Pillon B, , Yelnik J, & Mesnage V, et al. : Tourette's syndrome and deep brain stimulation. J Neurol Neurosurg Psychiatry 76:992995, 2005

    • Search Google Scholar
    • Export Citation
  • 25

    Jankovic J: Tourette syndrome. N Engl J Med 345:11841192, 2001

  • 26

    Jin R, , Zheng RY, & Huang WW, et al. : Epidemiological survey of Tourette syndrome in children and adolescents in Wenzhou of P.R. China. Eur J Epidemiol 20:925927, 2005

    • Search Google Scholar
    • Export Citation
  • 27

    Khalifa N, & von Knorring AL: Tourette syndrome and other tic disorders in a total population of children: clinical assessment and background. Acta Paediatr 94:16081614, 2005

    • Search Google Scholar
    • Export Citation
  • 28

    Leckman JF: Tourette's syndrome. Lancet 360:15771586, 2002

  • 29

    Leckman JF, , Riddle MA, , Hardin MT, , Ort SI, , Swartz JL, & Stevenson J, et al. : The Yale Global Tic Severity Scale: initial testing of a clinician-rated scale of tic severity. J Am Acad Child Adolesc Psychiatry 28:566573, 1989

    • Search Google Scholar
    • Export Citation
  • 30

    Leckman JF, , Zhang H, , Vitale A, , Lahnin F, , Lynch K, & Bondi C, et al. : Course of tic severity in Tourette syndrome: the first two decades. Pediatrics 102:1419, 1998

    • Search Google Scholar
    • Export Citation
  • 31

    Maddux BN, , Riley DE, , Whitney CM, & Maciunas RJ: Clinical efficacy and video analysis of deep brain stimulation for medically intractable Tourette Syndrome. Mov Disord 19:1123, 2004. (Abstract)

    • Search Google Scholar
    • Export Citation
  • 32

    Mink JW: Basal ganglia dysfunction in Tourette's syndrome: a new hypothesis. Pediatr Neurol 25:190198, 2001

  • 33

    Ozonoff S, & Jensen J: Brief report. Specific executive function profiles in three neurodevelopmental disorders. J Autism Dev Disord 29:171177, 1999

    • Search Google Scholar
    • Export Citation
  • 34

    Pappert EJ, , Goetz CG, , Louis ED, , Blasucci L, & Leurgans S: Objective assessments of longitudinal outcome in Gilles de la Tourette syndrome. Neurology 61:936940, 2003

    • Search Google Scholar
    • Export Citation
  • 35

    Robertson MM: Diagnosing Tourette syndrome. Is it a common disorder?. J Psychosom Res 55:36, 2003

  • 36

    Robertson MM: Tourette syndrome, associated conditions, and the complexities of treatment. Brain 123:425462, 2000

  • 37

    Shahed J, , Poysky J, , Kenney C, & Jankovic J: Bilateral GPi deep brain stimulation for Tourette Syndrome. Neurology 66:2 Suppl A366, 2006. (Abstract)

    • Search Google Scholar
    • Export Citation
  • 38

    Shucard DW, , Benedict RGB, , Tekok-Kilie A, & Lichter DG: Slowed reaction time during a continuous performance test in children with Tourette's syndrome. Neuropsychology 11:147155, 1997

    • Search Google Scholar
    • Export Citation
  • 39

    Silverstein SM, , Como PGG, , Palumbo DR, , West LL, & Osborn LM: Multiple sources of attentional dysfunction in adults with Tourette's syndrome. Neuropsychology 9:157164, 1995

    • Search Google Scholar
    • Export Citation
  • 40

    Temel Y, & Visser-Vandewalle V: Surgery in Tourette syndrome. Mov Disord 91:314, 2004

  • 41

    Torrance GW, , Feeny D, & Furlong W: Visual Analog Scales: do they have a role in the measurement of preferences for health states?. Med Decis Making 21:329334, 2001

    • Search Google Scholar
    • Export Citation
  • 42

    Visser-Vandewalle V, , Temel Y, , Boon P, , Vreeling F, , Colle H, & van der Linden C, et al. : Chronic bilateral thalamic stimulation: a new therapeutic approach in intractable Tourette syndrome. A report of three cases. J Neurosurg 99:10941100, 2003

    • Search Google Scholar
    • Export Citation
  • 43

    Ware JE, , Kosinski M, & Dewey JE: How to Score Version 2 of the SF-36 Health Survey Lincoln, RI, QualityMetric Incorporated, 2000

Metrics

All Time Past Year Past 30 Days
Abstract Views 1433 418 77
Full Text Views 332 31 0
PDF Downloads 225 30 0
EPUB Downloads 0 0 0