Secondary Worsening Following DYT1 Dystonia Deep Brain Stimulation: A Multi-country Cohort

Frontiers in Human Neuroscience
Takashi TsuboiMichael S Okun

Abstract

Objective: To reveal clinical characteristics of suboptimal responses to deep brain stimulation (DBS) in a multi-country DYT1 dystonia cohort. Methods: In this multi-country multi-center retrospective study, we analyzed the clinical data of DYT1 patients who experienced suboptimal responses to DBS defined as <30% improvement in dystonia scales at the last follow-up compared with baseline. We used a literature-driven historical cohort of 112 DYT1 patients for comparison. Results: Approximately 8% of our study cohort (11 out of 132) experienced suboptimal responses to DBS. Compared with the historical cohort, the multi-country cohort with suboptimal responses had a significantly younger age at onset (mean, 7.0 vs. 8.4 years; p = 0.025) and younger age at DBS (mean, 12.0 vs. 18.6 years; p = 0.019). Additionally, cranial involvement was more common in the multi-country cohort (before DBS, 64% vs. 45%, p = 0.074; before or after DBS, 91% vs. 47%, p = 0.001). Mean motor improvement at the last follow-up from baseline were 0% and 66% for the multi-country and historical cohorts, respectively. All 11 patients of the multi-country cohort had generalization of dystonia within 2.5 years after disease onset. All patients experienced dyston...Continue Reading

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Citations

Jan 26, 2021·Frontiers in Neurology·Stephen Tisch, Kishore Raj Kumar
Jun 3, 2021·Parkinsonism & Related Disorders·Eoin MulroyPatricia Limousin
Aug 28, 2021·The International Journal of Neuropsychopharmacology·Juan Carlos Benedetti-IsaacNorman López

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