Long-Term Clinical Outcome of Internal Globus Pallidus Deep Brain Stimulation for Dystonia

Park, Hye Ran; Lee, Jae Meen; Ehm, Gwanhee; Yang, Hui; Song, In Ho; Lim, Yong Beom; Kim, Mi Ryoung; Kim, Keyoung Ran; Lee, Woong-Woo; Kim, Young Eun; Hwang, Jae Ha; Shin, Chaewon; Park, Hye-Young; Kim, Jin Wook; Kim, Han Joon; Kim, Cheolyoung; Kim, Dong Gyu; Jeon, Beom S.; Paek, Sun Ha

doi:10.1371/journal.pone.0146644

Cited by 15 publications

(9 citation statements)

References 51 publications

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“…Previous studies have shown that both dystonia patients with DYT-1 and without a known genetic cause showed marked improvement after GPi-DBS. 36 Hence, future studies should control for this in larger populations.…”

Section: Discussionmentioning

confidence: 99%

Cortical network fingerprints predict deep brain stimulation outcome in dystonia

et al. 2019

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Background Deep brain stimulation (DBS) is an effective evidence‐based therapy for dystonia. However, no unequivocal predictors of therapy responses exist. We investigated whether patients optimally responding to DBS present distinct brain network organization and structural patterns. Methods From a German multicenter cohort of 82 dystonia patients with segmental and generalized dystonia who received DBS implantation in the globus pallidus internus, we classified patients based on the clinical response 3 years after DBS. Patients were assigned to the superior‐outcome group or moderate‐outcome group, depending on whether they had above or below 70% motor improvement, respectively. Fifty‐one patients met MRI‐quality and treatment response requirements (mean age, 51.3 ± 13.2 years; 25 female) and were included in further analysis. From preoperative MRI we assessed cortical thickness and structural covariance, which were then fed into network analysis using graph theory. We designed a support vector machine to classify subjects for the clinical response based on individual gray‐matter fingerprints. Results The moderate‐outcome group showed cortical atrophy mainly in the sensorimotor and visuomotor areas and disturbed network topology in these regions. The structural integrity of the cortical mantle explained about 45% of the DBS stimulation amplitude for optimal response in individual subjects. Classification analyses achieved up to 88% of accuracy using individual gray‐matter atrophy patterns to predict DBS outcomes. Conclusions The analysis of cortical integrity, informed by group‐level network properties, could be developed into independent predictors to identify dystonia patients who benefit from DBS. © 2019 The Authors. Movement Disorders published by Wiley Periodicals, Inc. on behalf of International Parkinson and Movement Disorder Society.

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Section: Discussionmentioning

confidence: 99%

Cortical network fingerprints predict deep brain stimulation outcome in dystonia

et al. 2019

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“…In adult patients, DBS targeting the globus pallidus interna (GPi) has already been successfully used in the management of dystonia. [5][6][7] The GPi is part of a cortico-striato-pallido-thalamo-cortical loop that has traditionally been implicated in the pathogenesis and pathophysiology of dystonia. 8 Several studies have also indicated that GPi-DBS can reduce the severity of dystonia in pediatric patients.…”

Section: Introductionmentioning

confidence: 99%

Subthalamic Nucleus Stimulation in Pediatric Isolated Dystonia: A 10-Year Follow-up

Zhang

et al. 2020

Can. J. Neurol. Sci.

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ABSTRACT:Objective:To evaluate the short-term and long-term clinical effectiveness and safety of subthalamic nucleus deep brain stimulation (STN-DBS) for medically intractable pediatric isolated dystonia.Methods:Using a longitudinal retrospective design, we assessed the clinical outcomes of nine patients who underwent STN-DBS for treatment-refractory pediatric isolated dystonia one decade ago (mean age at surgery: 15.9 ± 4.5 years). The primary clinical outcome used was assessed by retrospective video analyses of patients’ dystonia symptoms using the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS). Clinical assessments were performed at baseline, 1-year follow-up (1-yr FU), and 10-year follow-up (10-yr FU). Adverse side effects, including surgery-related, device-related, and stimulation-related effects, were also documented.Results:After STN-DBS surgery, the mean improvement in the BFMDRS motor score was 77.1 ± 26.6% at 1-yr FU and 90.4 ± 10.4% at 10-yr FU. Similarly, the mean BFMDRS disability score was improved by 69.5 ± 13.6% at 1-yr FU and by 86.5 ± 13.9% at 10-yr FU. The clinical improvements gained at 10-yr FU were significantly larger than those observed at 1-yr FU. Negative correlations were found between the duration of disease to age at surgery ratio (DD/AS) and the improvements in the BFMDRS motor score and total score at 1-yr FU and 10-yr FU.Conclusion:To our knowledge, this study provides the first clinical evidence for the short- and long-term effectiveness and safety of STN-DBS for pediatric isolated dystonia. Additionally, putative evidence is provided that earlier STN-DBS intervention in patients with refractory pediatric isolated dystonia may improve short- and long-term clinical outcomes.

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“…Additionally, the DYT-1 status has also been reported to be associated with treatment outcome[25] this distinction that was not made in our patients. Despite previous studies have shown that both, dystonia patients with DYT-1 and without a known genetic cause showed marked improvement after GPi-DBS,[26]. Future studies should control for this in larger populations.…”

Section: Discussionmentioning

confidence: 82%

Cortical network fingerprints predict deep brain stimulation outcome in dystonia

González‐Escamilla

Reich

Koirala

et al. 2018

Preprint

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Background: Deep brain stimulation (DBS) is an effective evidence-based therapy for dystonia. However, no unequivocal predictors of therapy responses exist. We investigate whether patients optimally responding to DBS present distinct brain network organization and structural patterns.Methods: Based on a German multicentre cohort of eighty-two dystonia patients with segmental and generalized dystonia, who received DBS implantation in the globus pallidus internus patients were classified based on the clinical response 36 months after DBS, as superior-outcome group or moderate-outcome group, as above or below 70% motor improvement, respectively. Fifty-one patients met MRI-quality and treatment response requirements (mean age 51.3 ± 13.2 years; 25 female) and were included into further analysis. From preoperative MRI we assessed cortical thickness and structural covariance, which were then fed into network analysis using graph theory. We designed a support vector machine to classify subjects for the clinical response based on group network properties and individual grey matter fingerprints. Results:The moderate-outcome group showed cortical atrophy mainly in the sensorimotor and visuomotor areas and disturbed network topology in these regions. From all the structural integrity of the cortical mantle explained about 45% of the stimulation amplitude.Classification analyses achieved 88% of accuracy using individual grey matter atrophy patterns to predict DBS outcome. Conclusions:The analysis of cortical integrity and network properties could be developed into independent predictors to identify dystonia patients who benefit from DBS.

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Long-Term Clinical Outcome of Internal Globus Pallidus Deep Brain Stimulation for Dystonia

Cited by 15 publications

References 51 publications

Cortical network fingerprints predict deep brain stimulation outcome in dystonia

Cortical network fingerprints predict deep brain stimulation outcome in dystonia

Subthalamic Nucleus Stimulation in Pediatric Isolated Dystonia: A 10-Year Follow-up

Cortical network fingerprints predict deep brain stimulation outcome in dystonia

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