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Sun Ha Paek 4 Articles
Long-Term Outcomes of Deep Brain Stimulation in Pantothenate Kinase-Associated Neurodegeneration-Related Dystonia
Kyung Ah Woo, Han-Joon Kim, Seung-Ho Jeon, Hye Ran Park, Kye Won Park, Seung Hyun Lee, Sun Ju Chung, Jong-Hee Chae, Sun Ha Paek, Beomseok Jeon
J Mov Disord. 2022;15(3):241-248.   Published online July 26, 2022
  • 3,267 View
  • 178 Download
  • 3 Web of Science
  • 4 Crossref
AbstractAbstract PDFSupplementary Material
To investigate the long-term clinical outcomes of pallidal deep brain stimulation (GPi-DBS) in patients with pantothenate kinase-associated neurodegeneration (PKAN).
We reviewed the records of patients with genetically confirmed PKAN who received bilateral GPi-DBS for refractory dystonia and were clinically followed up for at least 2 years postoperatively at two centers in Korea. Pre- and postoperative Burke– Fahn–Marsden Dystonia Rating Scale motor subscale (BFMDRS-M) scores, disability subscale (BFMDRS-D) scores, and qualitative clinical information were prospectively collected. Descriptive analysis was performed for BFMDRS-M scores, BFMDRSD scores, and the orofacial, axial, and limb subscores of the BFMDRS-M at 6–12, 24–36, and 60–72 months postoperatively.
Five classic-type, four atypical-type, and one unknown-type PKAN cases were identified. The mean preoperative BFMDRS-M score was 92.1 for the classic type and 38.5 for the atypical or unknown type, with a mean BFMDRS follow-up of 50.7 months and a clinical follow-up of 69.0 months. The mean improvements in BFMDRS-M score were 11.3%, 41.3%, and 30.5% at 6–12, 24–36, and 60–72 months, respectively. In four patients with full regular evaluations until 60–72 months, improvements in the orofacial, axial, and limb subscores persisted, but the disability scores worsened from 24–36 months post-operation compared to the baseline, mainly owing to the aggravation of eating and feeding disabilities.
The benefits of GPi-DBS on dystonia may persist for more than 5 years in PKAN. The effects on patients’ subjective disability may have a shorter duration despite improvements in dystonia owing to the complex manifestations of PKAN.


Citations to this article as recorded by  
  • Deep Brain Stimulation for Refractory Status Dystonicus in Children: Multicenter Case Series and Systematic Review
    Lindsey M. Vogt, Han Yan, Brendan Santyr, Sara Breitbart, Melanie Anderson, Jürgen Germann, Karlo J. Lizarraga, Angela L. Hewitt, Alfonso Fasano, George M. Ibrahim, Carolina Gorodetsky
    Annals of Neurology.2024; 95(1): 156.     CrossRef
  • Illustration of the long-term efficacy of pallidal deep brain stimulation in a patient with PKAN dystonia
    Luigi M. Romito, Fabiana Colucci, Giovanna Zorzi, Barbara Garavaglia, Ahmet Kaymak, Alberto Mazzoni, Celeste Panteghini, Nico Golfrè Andreasi, Sara Rinaldo, Vincenzo Levi, Miryam Carecchio, Roberto Eleopra
    Parkinsonism & Related Disorders.2024; 123: 106977.     CrossRef
  • Case of Hallervorden–Spatz Syndrome: A Tale of Twin Sisters
    Naveen Reddy, Jitender Sharma, Anmol Sharma
    Neurology India.2024; 72(2): 411.     CrossRef
  • Surgical treatment of movement disorders in neurometabolic conditions
    Alonso Zea Vera, Andrea L. Gropman
    Frontiers in Neurology.2023;[Epub]     CrossRef
Development of Clinical Milestones in Parkinson’s Disease After Bilateral Subthalamic Deep Brain Stimulation
Jed Noel A. Ong, Jung Hwan Shin, Seungho Jeon, Chan Young Lee, Han-Joon Kim, Sun Ha Paek, Beomseok Jeon
J Mov Disord. 2022;15(2):124-131.   Published online May 26, 2022
  • 2,710 View
  • 143 Download
  • 1 Web of Science
  • 1 Crossref
AbstractAbstract PDFSupplementary Material
Deep brain stimulation of the subthalamic nucleus (STN-DBS) in Parkinson’s disease (PD) patients does not halt disease progression, as these patients will progress and develop disabling non-levodopa responsive symptoms. These features may act as milestones that represent the overall functionality of patients after DBS. The objective of this study was to investigate the development of clinical milestones in advanced PD patients who underwent bilateral STN-DBS.
The study evaluated PD patients who underwent STN-DBS at baseline up to their last follow-up using the Unified Parkinson’s Disease Rating Scale and Hoehn and Yahr scale. The symptoms of hallucinations, dysarthria, dysphagia, frequent falls, difficulty walking, cognitive impairment and the loss of autonomy were chosen as the clinical milestones.
A total of 106 patients with a mean age of 47.21 ± 10.52 years at disease onset, a mean age of 58.72 ± 8.74 years at surgery and a mean disease duration of 11.51 ± 4.4 years before surgery were included. Initial improvement of motor symptoms was seen after the surgery with the appearance of clinical milestones over time. Using the moderately disabling criteria, 81 patients (76.41%) developed at least one clinical milestone, while 48 patients (45.28%) developed a milestone when using the severely disabling criteria.
STN-DBS has a limited effect on axial and nonmotor symptoms of the PD patients, in contrast to the effect on motor symptoms. These symptoms may serve as clinical milestones that can convey the status of PD patients and its impact on the patients and their caregivers. Therefore, advanced PD patients, even those treated with bilateral STN-DBS, will still require assistance and cannot live independently in the long run.


Citations to this article as recorded by  
  • Unveiling the Impact of Outpatient Physiotherapy on Specific Motor Symptoms in Parkinson’s Disease: A Prospective Cohort Study
    Yuta Terasawa, Koki Ikuno, Shintaro Fujii, Yuki Nishi, Emi Tanizawa, Sachio Nabeshima, Yohei Okada
    Brain & Neurorehabilitation.2023;[Epub]     CrossRef
Successful Pallidal Deep Brain Stimulation in a Patient with Childhood-Onset Generalized Dystonia with ANO3 Mutation
Dallah Yoo, Han-Joon Kim, Jong-Hee Chae, Sun Ha Paek, Beomseok Jeon
J Mov Disord. 2019;12(3):190-191.   Published online July 17, 2019
  • 5,510 View
  • 122 Download
  • 9 Web of Science
  • 9 Crossref
PDFSupplementary Material


Citations to this article as recorded by  
  • The Clinical Spectrum of ANO3—Report of a New Family and Literature Review
    Marco Percetti, Michela Zini, Paola Soliveri, Filippo Cogiamanian, Mariarosa Ferrara, Eva Orunesu, Alessandra Ranghetti, Carlo Ferrarese, Gianni Pezzoli, Barbara Garavaglia, Ioannis Ugo Isaias, Giorgio Sacilotto
    Movement Disorders Clinical Practice.2024; 11(3): 289.     CrossRef
  • The role of genetics in the treatment of dystonia with deep brain stimulation: Systematic review and Meta-analysis
    Harini Sarva, Federico Rodriguez-Porcel, Francisco Rivera, Claudio Daniel Gonzalez, Samantha Barkan, Susmit Tripathi, Emilia Gatto, Pedro Garcia Ruiz
    Journal of the Neurological Sciences.2024; 459: 122970.     CrossRef
  • A novel ANO3 variant in two siblings with different phenotypes
    Marcello Esposito, Assunta Trinchillo, Francesca Piceci-Sparascio, Maria Cecilia D'Asdia, Federica Consoli, Alessandro De Luca
    Parkinsonism & Related Disorders.2023; 111: 105413.     CrossRef
  • DBS-Evoked Pallidal Activity Correlates with Clinical Improvement in a Patient with ANO3-Related Dystonia
    Aditya Boddu, Adam Bashir, Mohammad Awad, Barton Guthrie, Harrison Walker
    SSRN Electronic Journal.2023;[Epub]     CrossRef
  • Deep Brain Stimulation in Dystonia: Disentangling Heterogeneity
    Alberto Albanese
    Movement Disorders Clinical Practice.2021; 8(1): 6.     CrossRef
  • Pallidal Deep Brain Stimulation for Monogenic Dystonia: The Effect of Gene on Outcome
    Stephen Tisch, Kishore Raj Kumar
    Frontiers in Neurology.2021;[Epub]     CrossRef
  • The expanding clinical and genetic spectrum of ANO3 dystonia
    Li-Ting Jiang, Li-Xi Li, Ying Liu, Xiao-Long Zhang, You-Gui Pan, Lin Wang, Xin-Hua Wan, Ling-Jing Jin
    Neuroscience Letters.2021; 746: 135590.     CrossRef
  • Huntington disease-like phenotype in a patient with ANO3 mutation
    Shahedah Koya Kutty, Eoin Mulroy, Francesca Magrinelli, Giulia Di Lazzaro, Anna Latorre, Kailash P. Bhatia
    Parkinsonism & Related Disorders.2021; 90: 120.     CrossRef
  • Arching deep brain stimulation in dystonia types
    Han-Joon Kim, Beomseok Jeon
    Journal of Neural Transmission.2021; 128(4): 539.     CrossRef
Nonmotor Symptoms and Subthalamic Deep Brain Stimulation in Parkinson’s Disease
Han-Joon Kim, Beom S. Jeon, Sun Ha Paek
J Mov Disord. 2015;8(2):83-91.   Published online May 31, 2015
  • 24,982 View
  • 248 Download
  • 55 Web of Science
  • 47 Crossref
AbstractAbstract PDF
Subthalamic deep brain stimulation (STN DBS) is an established treatment for the motor symptoms in patients with advanced Parkinson’s disease (PD). In addition to improvements in motor symptoms, many studies have reported changes in various nonmotor symptoms (NMSs) after STN DBS in patients with PD. Psychiatric symptoms, including depression, apathy, anxiety, and impulsivity, can worsen or improve depending on the electrical stimulation parameters, the locations of the stimulating contacts within the STN, and changes in medications after surgery. Global cognitive function is not affected by STN DBS, and there is no increase in the incidence of dementia after STN DBS compared to that after medical treatment, although clinically insignificant declines in verbal fluency have been consistently reported. Pain, especially PD-related pain, improves with STN DBS. Evidence regarding the effects of STN DBS on autonomic symptoms and sleep-related problems is limited and remains conflicting. Many symptoms of nonmotor fluctuations, which are occasionally more troublesome than motor fluctuations, improve with STN DBS. Although it is clear that NMSs are not target symptoms for STN DBS, NMSs have a strong influence on the quality of life of patients with PD, and clinicians should thus be aware of these NMSs when deciding whether to perform surgery and should pay attention to changes in these symptoms after STN DBS to ensure the optimal care for patients.


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    Parkinson's Disease.2024; 2024: 1.     CrossRef
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    The American Journal of Geriatric Psychiatry.2023;[Epub]     CrossRef
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    G. Brand, C. Bontempi, L. Jacquot
    Revue Neurologique.2023; 179(9): 947.     CrossRef
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    Naeem Muhammad, Saurabh Sonkusare, Qiong Ding, Linbin Wang, Alekhya Mandali, Yi Jie Zhao, Bomin Sun, Dianyou Li, Valerie Voon
    Frontiers in Human Neuroscience.2023;[Epub]     CrossRef
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    Current Nutrition & Food Science.2023; 19(9): 961.     CrossRef
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    Movement Disorders Clinical Practice.2022; 9(1): 48.     CrossRef
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    Zahra Eghlidos, Zahra Rahimian, Gholamreza Vadiee, Soodeh Jahangiri
    Acta Neurologica Scandinavica.2022; 146(2): 115.     CrossRef
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    Bowen Chang, Chen Ni, Jiaming Mei, Chi Xiong, Peng Chen, Manli Jiang, Chaoshi Niu
    Brain Sciences.2022; 12(7): 841.     CrossRef
  • A nigra–subthalamic circuit is involved in acute and chronic pain states
    Cui Yin, Tao Jia, Yiwen Luan, Xue Zhang, Cheng Xiao, Chunyi Zhou
    Pain.2022; 163(10): 1952.     CrossRef
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    Tao Jia, Ying-Di Wang, Jing Chen, Xue Zhang, Jun-Li Cao, Cheng Xiao, Chunyi Zhou
    Nature Communications.2022;[Epub]     CrossRef
  • Basal impulses: findings from the last twenty years on impulsivity and reward pathways using deep brain stimulation
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    Journal of Neurosurgical Sciences.2021;[Epub]     CrossRef
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    Jiayu Liu, Hu Ding, Ke Xu, Ruen Liu, Dongliang Wang, Jia Ouyang, Zhi Liu, Zeyu Miao
    Scientific Reports.2021;[Epub]     CrossRef
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    Proceedings of the National Academy of Sciences.2020; 117(18): 10045.     CrossRef
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    Sara Scherrer, Andrew H. Smith, Jaimie Gowatsky, Christina A. Palmese, Joohi Jimenez-Shahed, Brian H. Kopell, Helen S. Mayberg, Martijn Figee
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    Hye Ran Park, Yong Hoon Lim, Eun Jin Song, Jae Meen Lee, Kawngwoo Park, Kwang Hyon Park, Woong-Woo Lee, Han-Joon Kim, Beomseok Jeon, Sun Ha Paek
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    M. Acera, A. Molano, B. Tijero, G. Bilbao, I. Lambarri, R. Villoria, J. Somme, E. Ruiz de Gopegui, I. Gabilondo, J.C. Gomez-Esteban
    Neurología (English Edition).2019; 34(9): 573.     CrossRef
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    Maryam Ghahremani, Jaejun Yoo, Sun Ju Chung, Kwangsun Yoo, Jong C. Ye, Yong Jeong
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    J.A. Crespo-Burillo, D. Rivero-Celada, A. Saenz-de Cabezón, J. Casado-Pellejero, J. Alberdi-Viñas, R. Alarcia-Alejos
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    Ying Wang, Yongsheng Li, Xiaona Zhang, Anmu Xie
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    Clinical Neurology and Neurosurgery.2018; 174: 174.     CrossRef
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    Acta Neurologica Scandinavica.2018;[Epub]     CrossRef
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