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

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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.

Article Information

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.

© AANS, except where prohibited by US copyright law.

Headings

Figures

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    Timeline representing steps in the pilot study. Video camera icons designate video recording sessions. Assessment of secondary outcome measures are indicated by N (neuropsychological battery), Q (quality of life), T (TSSL), and Y (YGTSS). Combins = combinations; f/u = follow-up.

  • View in gallery

    Coronal and axial T1-weighted MR images featuring the target point in all five surgically treated patients. A, D, and E: Responders (Cases 1, 4, and 5). B and C: Nonresponders (Cases 2 and 3). The image labeled “Plan” (lower right) is in the oblique plane, along electrodes, of the planned thalamic target and electrode trajectory using the BrainLab software.

  • View in gallery

    Plots depicting motor (A and C) and sonic (B and D) tic counts normalized so that the preoperative count equals 1. The cross-hairs indicate the 50th percentile; the upper and lower box limits, the 25th and 75th percentiles; and the vertical span, the 0 and 100th percentiles. L = left; pre-stim = prestimulation; R = right.

  • View in gallery

    Plots revealing secondary outcome measures at the preoperative and 3-month follow-up assessments. A: Complete-scale YGTSS scores. B: The VAS scores. C: The SF-36 scores. The cross-hairs indicate the 50th percentile; the upper and lower box limits, the 25th and 75th percentiles; and the vertical span, the 0 and 100th percentiles. QoL = quality of life.

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