Journal of Movement Disorders

Original Article

Optimal Stimulation Sites and Long-Term Efficacy of Pallidal Deep-Brain Stimulation for Patients With Tardive Dystonia: Taku Nonaka, Shiro Horisawa, Kilsoo Kim, Masato Murakami, Masahiko Nishitani, Takakazu Kawamata, Takaomi Taira; J Mov Disord. 2026;19(1):49-57. Published online October 28, 2025; DOI: https://doi.org/10.14802/jmd.25164

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Abstract PDF: Objective
Globus pallidus internus deep brain stimulation (GPi-DBS) is an established treatment for dystonia, but its specific efficacy for tardive dystonia (TD) remains insufficiently documented. To evaluate the long-term clinical outcomes of GPi-DBS and to identify optimal stimulation sites in patients with medically refractory TD.
Methods
We retrospectively analyzed data from 26 patients with TD who underwent bilateral GPi-DBS. Clinical outcomes were assessed using the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS). Optimal stimulation sites were identified using voxelwise sweet spot analysis.
Results
At an average follow-up time of 42 months (range 12–4 months), the mean BFMDRS score improvement was 81.5%. The optimal stimulation sites were located in the posteroventral region of the GPi. Two patients experienced sustained symptom remission after DBS cessation. Complications included device-related infection (n=2), dysarthria (n=4), and gait imbalance (n= 1); no severe permanent complications occurred.
Conclusion
GPi-DBS is effective and safe for patients with medically refractory TD, providing significant long-term symptom relief. The optimal stimulation sites were located in the posteroventral GPi, which is consistent with those reported for patients with other dystonia types.

Review Article

Pallidus Stimulation for Chorea-Acanthocytosis: A Systematic Review and Meta-Analysis of Individual Data: Weibin He, Chenhui Li, Hongjuan Dong, Lingmin Shao, Bo Yin, Dianyou Li, Liguo Ye, Ping Hu, Chencheng Zhang, Wei Yi; J Mov Disord. 2022;15(3):197-205. Published online July 26, 2022; DOI: https://doi.org/10.14802/jmd.22003

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A significant proportion of patients with chorea-acanthocytosis (ChAc) fail to respond to standard therapies. Recent evidence suggests that globus pallidus internus (GPi) deep brain stimulation (DBS) is a promising treatment option; however, reports are few and limited by sample sizes. We conducted a systematic literature review to evaluate the clinical outcome of GPi-DBS for ChAc. PubMed, Embase, and Cochrane Library databases were searched for relevant articles published before August 2021. The improvement of multiple motor and nonmotor symptoms was qualitatively presented. Improvements in the Unified Huntington’s Disease Rating Scale motor score (UHDRS-MS) were also analyzed during different follow-up periods. A multivariate linear regression analysis was conducted to identify potential predictors of clinical outcomes. Twenty articles, including 27 patients, were eligible. Ninety-six percent of patients with oromandibular dystonia reported significant improvement. GPi-DBS significantly improved the UHDRS-motor score at < 6 months (p < 0.001) and ≥ 6 months (p < 0.001). The UHDRS-motor score improvement rate was over 25% in 75% (15/20 cases) of patients at long-term follow-up (≥ 6 months). The multiple linear regression analysis showed that sex, age at onset, course of disease, and preoperative movement score had no linear relationship with motor improvement at long-term follow-up (p > 0.05). GPi-DBS is an effective and safe treatment in most patients with ChAc, but no reliable predictor of efficacy has been found. Oromandibular dystonia-dominant patients might be the best candidates for GPi-DBS.

Citations

Citations to this article as recorded by

Subthalamic Deep Brain Stimulation for Chorea-Acanthocytosis: A Single-Center Case Series
Tianyu Ma, Meitong Zhou, Suzhen Lin, Tao Wang, Peng Huang, Dianyou Li, Hongxia Li, Yiwen Wu
Neuromodulation: Technology at the Neural Interface.2026;[Epub] CrossRef
Chorea-acanthocytosis presenting with epilepsy at onset harboring novel compound heterozygous VPS13A mutations
Mitsuyoshi Tamura, Shogo Furukawa, Masahiro Namiki, Yukiko Ozawa, Yuka Urata, Masayuki Nakamura, Mitsuru Watanabe, Hideki Oshima, Toshiki Obuchi, Tadashi Ichikawa, Yuriko Kikkawa
Clinical Parkinsonism & Related Disorders.2026; 14: 100433. CrossRef
38-jähriger Patient mit Epilepsie, hyperkinetischer Bewegungsstörung und Hyper-CKämie
E. Fischer, A. Großmann, H. Ludwig, M. Lanz, A. Mühlbäck, A. Hermann, K. Peikert
DGNeurologie.2026; 9(3): 221. CrossRef
Immunology of familial chorea-acanthocytosis with presenting generalized tonic-clonic seizure: Blood cell study for early diagnosis and management
Amineh Salem, Narges Omidvar
Cellular Immunology.2025; 411-412: 104946. CrossRef
Clinical neurophysiology in the treatment of movement disorders: IFCN handbook chapter
Jean-Pascal Lefaucheur, Elena Moro, Yuichiro Shirota, Yoshikazu Ugawa, Talyta Grippe, Robert Chen, David H Benninger, Bahman Jabbari, Sanaz Attaripour, Mark Hallett, Walter Paulus
Clinical Neurophysiology.2024; 164: 57. CrossRef
Bilateral deep brain stimulation (DBS) of globus pallidus internus (GPi) for the treatment of benign hereditary chorea and other childhood onset choreas: a single-center experience
Arthur R. Kurzbuch, Ben Cooper, Gina Lumsdon, Nicola Idowu, Helen Gedrim, Philipa Mulholland, Volker Tronnier, Ram Kumar, Jonathan R. Ellenbogen
Neurosurgical Review.2024;[Epub] CrossRef

Case Report

Holmes’ Tremor with Shoulder Pain Treated by Deep Brain Stimulation of Unilateral Ventral Intermediate Thalamic Nucleus and Globus Pallidus Internus: Sabri Aydın, Huseyin Canaz, Ezgi Tuna Erdogan, Nazlı Durmaz, Barıs Topcular; J Mov Disord. 2017;10(2):92-95. Published online April 18, 2017; DOI: https://doi.org/10.14802/jmd.16051

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Abstract PDF

A 21-year-old male was admitted with severe right arm and hand tremors after a thalamic hemorrhage caused by a traffic accident. He was also suffering from agonizing pain in his right shoulder that manifested after the tremor. Neurologic examination revealed a disabling, severe, and irregular kinetic and postural tremor in the right arm during target-directed movements. There was also an irregular ipsilateral rest tremor and dystonic movements in the distal part of the right arm. The amplitude was moderate at rest and extremely high during kinetic and intentional movements. The patient underwent left globus pallidum internus and ventral intermediate thalamic nucleus deep brain stimulation. The patient improved by more than 80% as rated by the Fahn-Tolosa-Marin Tremor Rating Scale and Visual Analog Scale six months after surgery.

Citations

Citations to this article as recorded by

Efficacy and safety of multiple-target deep brain stimulation in non-parkinsonian movement disorders: a systematic review
Evangelos Kalogirou, Spyridon Voulgaris, George A Alexiou
Neurosurgical Review.2025;[Epub] CrossRef
Holmes tremor: an updated review
Efstratios-Stylianos Pyrgelis, Eleni Agapiou, Efthalia Angelopoulou
Neurological Sciences.2022; 43(12): 6731. CrossRef
Deep brain stimulation of the posterior subthalamic area as an alternative strategy for management of Holmes tremor: A case report and review of the literature
Omid Yousefi, Mojtaba Dayyani, Razieh Rezaei, Hooman Kamran, Ali Razmkon
Surgical Neurology International.2022; 13: 489. CrossRef
Vim stereotactic radiosurgical thalamotomy for drug-resistant idiopathic Holmes tremor: a case report
Manjul Tripathi, Sahil Mehta, Raghav Singla, Chirag K. Ahuja, Naresh Tandalya, Constantin Tuleasca, Aman Batish, Sandeep Mohindra, Abhinav Agrahari, Rupinder Kaur
Acta Neurochirurgica.2021; 163(7): 1867. CrossRef
Deep brain stimulation in uncommon tremor disorders: indications, targets, and programming
Carlo Alberto Artusi, Ashar Farooqi, Alberto Romagnolo, Luca Marsili, Roberta Balestrino, Leonard L. Sokol, Lily L. Wang, Maurizio Zibetti, Andrew P. Duker, George T. Mandybur, Leonardo Lopiano, Aristide Merola
Journal of Neurology.2018; 265(11): 2473. CrossRef

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