Vertebral artery dissection leading to stroke caused by violent neck tics of Tourette syndrome
Division of Neurology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, USA.Neurology (Impact Factor: 8.29). 11/2011; 77(18):1706-8. DOI: 10.1212/WNL.0b013e318238253c
Article: A ticcing time bomb?
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ABSTRACT: OBJECTIVE To collect the information necessary to design the methods and outcome variables for a larger trial of scheduled deep brain stimulation (DBS) for Tourette syndrome. DESIGN We performed a small National Institutes of Health-sponsored clinical trials planning study of the safety and preliminary efficacy of implanted DBS in the bilateral centromedian thalamic region. The study used a cranially contained constant-current device and a scheduled, rather than the classic continuous, DBS paradigm. Baseline vs 6-month outcomes were collected and analyzed. In addition, we compared acute scheduled vs acute continuous vs off DBS. SETTING A university movement disorders center. PATIENTS Five patients with implanted DBS. MAIN OUTCOME MEASURE A 50% improvement in the Yale Global Tic Severity Scale (YGTSS) total score. RESULTS Participating subjects had a mean age of 34.4 (range, 28-39) years and a mean disease duration of 28.8 years. No significant adverse events or hardware-related issues occurred. Baseline vs 6-month data revealed that reductions in the YGTSS total score did not achieve the prestudy criterion of a 50% improvement in the YGTSS total score on scheduled stimulation settings. However, statistically significant improvements were observed in the YGTSS total score (mean [SD] change, -17.8 [9.4]; P = .01), impairment score (-11.3 [5.0]; P = .007), and motor score (-2.8 [2.2]; P = .045); the Modified Rush Tic Rating Scale Score total score (-5.8 [2.9]; P = .01); and the phonic tic severity score (-2.2 [2.6]; P = .04). Continuous, off, and scheduled stimulation conditions were assessed blindly in an acute experiment at 6 months after implantation. The scores in all 3 conditions showed a trend for improvement. Trends for improvement also occurred with continuous and scheduled conditions performing better than the off condition. Tic suppression was commonly seen at ventral (deep) contacts, and programming settings resulting in tic suppression were commonly associated with a subjective feeling of calmness. CONCLUSIONS This study provides safety and proof of concept that a scheduled DBS approach could improve motor and vocal tics in Tourette syndrome. Refinements in neurostimulator battery life, outcome measure selection, and flexibility in programming settings can be used to enhance outcomes in a future larger study. Scheduled stimulation holds promise as a potential first step for shifting movement and neuropsychiatric disorders toward more responsive neuromodulation approaches. TRIAL REGISTRATION clinicaltrials.gov Identifier: NCT01329198.
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ABSTRACT: Pharmacological treatment is usually indicated in moderate to severe tics with according psychosocial and/or functional impairment. Neuroleptics with D2 antagonistic activity remain the cornerstone of anti-tic therapy. Lack of randomized controlled clinical trials base therapeutic decisions mainly on clinical expertise and common sense. Recently, aripiprazole has emerged as the neuroleptic with the most advantageous efficacy/side effect ratio for treating tics. Yet, in non-responders to aripiprazole, many neuroleptic and non-neuroleptic drugs, including botulinum toxin injections, are available and often successful. Apart from conducting methodologically sound trials (which includes sufficiently long observation periods), future efforts in the field should test the combination of cognitive-behavioural therapy with drugs or of multi-drug therapy as well as the development of biomarkers (endephenotypes) to monitor and even predict treatment response.
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