Long-term follow-up of DYT1 dystonia patients treated by deep brain stimulation: an open-label study

Fuente: arXiv
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Main Authors: Cif, Laura, Vasques, Xavier, Gonzalez, Victoria, Ravel, Patrice, Biolsi, Brigitte, Collod-Beroud, Gwenaelle, Tuffery-Giraud, Sylvie, Elfertit, Hassan, Claustres, Mireille, Coubes, Philippe
Format: Preprint
Published: 2025
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author Cif, Laura
Vasques, Xavier
Gonzalez, Victoria
Ravel, Patrice
Biolsi, Brigitte
Collod-Beroud, Gwenaelle
Tuffery-Giraud, Sylvie
Elfertit, Hassan
Claustres, Mireille
Coubes, Philippe
author_facet Cif, Laura
Vasques, Xavier
Gonzalez, Victoria
Ravel, Patrice
Biolsi, Brigitte
Collod-Beroud, Gwenaelle
Tuffery-Giraud, Sylvie
Elfertit, Hassan
Claustres, Mireille
Coubes, Philippe
contents Long-term efficacy of internal globus pallidus (GPi) deep-brain stimulation (DBS) in DYT1 dystonia and disease progression under DBS was studied. Twenty-six patients of this open-label study were divided into two groups: (A) with single bilateral GPi lead, (B) with a second bilateral GPi lead implanted owning to subsequent worsening of symptomatology. Dystonia was assessed with the Burke Scale. Appearance of new symptoms and distribution according to body region were recorded. In the whole cohort, significant decreases in motor and disability subscores (P < 0.0001) were observed at 1 year and maintained up to 10 years. Group B showed worsening of the symptoms. At 1 year, there were no significant differences between Groups A (without subsequent worsening) and B; at 5 years, a significant difference was found for motor and disability scores. Within Group B, four patients exhibited additional improvement after the second DBS surgery. In the 26 patients, significant difference (P = 0.001) was found between the number of body regions affected by dystonia preoperatively and over the whole follow-up. DBS efficacy in DYT1 dystonia can be maintained up to 10 years (two patients). New symptoms appear with long-term follow-up and may improve with additional leads in a subgroup of patients.
format Preprint
id arxiv_https___arxiv_org_abs_2502_13661
institution arXiv
publishDate 2025
record_format arxiv
spellingShingle Long-term follow-up of DYT1 dystonia patients treated by deep brain stimulation: an open-label study
Cif, Laura
Vasques, Xavier
Gonzalez, Victoria
Ravel, Patrice
Biolsi, Brigitte
Collod-Beroud, Gwenaelle
Tuffery-Giraud, Sylvie
Elfertit, Hassan
Claustres, Mireille
Coubes, Philippe
Neurons and Cognition
Long-term efficacy of internal globus pallidus (GPi) deep-brain stimulation (DBS) in DYT1 dystonia and disease progression under DBS was studied. Twenty-six patients of this open-label study were divided into two groups: (A) with single bilateral GPi lead, (B) with a second bilateral GPi lead implanted owning to subsequent worsening of symptomatology. Dystonia was assessed with the Burke Scale. Appearance of new symptoms and distribution according to body region were recorded. In the whole cohort, significant decreases in motor and disability subscores (P < 0.0001) were observed at 1 year and maintained up to 10 years. Group B showed worsening of the symptoms. At 1 year, there were no significant differences between Groups A (without subsequent worsening) and B; at 5 years, a significant difference was found for motor and disability scores. Within Group B, four patients exhibited additional improvement after the second DBS surgery. In the 26 patients, significant difference (P = 0.001) was found between the number of body regions affected by dystonia preoperatively and over the whole follow-up. DBS efficacy in DYT1 dystonia can be maintained up to 10 years (two patients). New symptoms appear with long-term follow-up and may improve with additional leads in a subgroup of patients.
title Long-term follow-up of DYT1 dystonia patients treated by deep brain stimulation: an open-label study
topic Neurons and Cognition
url https://arxiv.org/abs/2502.13661