Objective: We report the serendipity case of overnight stimulation for the management of stimulation-induced bradykinesia in two patients with generalized dystonia and clinically-effective pallidal deep brain stimulation.

Background: Generalized dystonia is characterized by involuntary muscle contractions that affect multiple muscle groups throughout the body and lead to progressive motor impairment. Deep brain stimulation of the Globus Pallidus (GPi-DBS) is an effective treatment for severe, medically refractory dystonia that can lead to sustained improvement of motor symptoms and quality of life. However, up to 8% of the patients effectively treated with GPi-DBS can develop bradykinesia with worsening of daily living activities. The management of this side-effect is challenging and limited by its unclear pathophysiology.

Method: We report two cases of early-onset generalized dystonia (P1: isolated idiopathic dystonia, male, caucasian, 37 years; P2: THAP1-dystonia, female, caucasian, 39 years) with a predominantly dystonic tremor in P1 and trunk-dystonia in P2 (Burke-Fahn-Marsden Dystonia Rating Scale, BFMRDS-P1: 8, -P2: 29 points). After bilateral GPi-DBS, dystonia improved in both subjects (BFMRDS-P1: 3, -P2: 3,5 points), but they developed bradykinesia with impairment of working activities. Extensive reprogramming could not improve bradykinesia without the re-emergence of dystonia. After excluding an electrode misplacement, we gave the patients the possibility of varying the DBS parameters greatly. They developed independently a novel paradigm of stimulation with high-intensity stimulation overnight and washout during the day.

Results: Overnight stimulation showed sustained improvement of dystonia over the day (BFMRDS-P1: 4, -P2: 4,5 points) without the development of bradykinesia and improved daily living activities. The different timing of clinical- and side-effect of GPi-DBS hints on possible distinct underlying mechanisms.

Conclusion: Overnight stimulation may be an effective rescue strategy for GPi-DBS-induced bradykinesia in dystonia.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/overnight-stimulation-a-new-rescue-strategy-for-deep-brain-stimulation-induced-bradykinesia-in-dystonia/