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Research Articles, Neurobiology of Disease

The Pathophysiology of Dystonic Tremors and Comparison With Essential Tremor

Pattamon Panyakaew, Hyun Joo Cho, Sang Wook Lee, Tianxia Wu and Mark Hallett
Journal of Neuroscience 25 November 2020, 40 (48) 9317-9326; DOI: https://doi.org/10.1523/JNEUROSCI.1181-20.2020
Pattamon Panyakaew
1Human Motor Control Section, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland 20892-1428
2Chulalongkorn Center of Excellence for Parkinson's Disease & Related Disorders, Department of Medicine, Faculty of Medicine, Chulalongkorn University and King Chulalongkorn Memorial Hospital, Thai Red Cross Society, Bangkok, 10330, Thailand
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Hyun Joo Cho
1Human Motor Control Section, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland 20892-1428
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Sang Wook Lee
3Department of Biomedical Engineering, Catholic University of America, Washington, DC 20064
4Center for Applied Biomechanics and Rehabilitation Research, MedStar National Rehabilitation Hospital, Washington, DC 20010
5Department of Mechanical Engineering, Korea Advanced Institute of Science and Technology, Daejeon, 34141, Korea
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Tianxia Wu
6Clinical Neurosciences Program, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland 20892-1428
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Mark Hallett
1Human Motor Control Section, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland 20892-1428
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Abstract

There are two types of dystonic tremor syndromes (DTS), dystonic tremor (DT) and tremor associated with dystonia (TAWD), and neither is understood. DTS likely share some mechanisms with nontremulous dystonia, and there may also be overlaps with essential tremor (ET). We studied 21 ET (8 females, 13 males) and 22 DTS human patients (10 females, 12 males), including 13 human patients with DT (writer's cramp with writing tremor) and 9 human patients with tremor associated with dystonia (TAWD; cervical dystonia with hand tremor). Tremors were analyzed using accelerometry and surface EMG of the antagonist pairs of arm muscles during posture, simple kinetic movement, and writing. Cerebellar inhibition was performed to assess cerebello-thalamo-cortical involvement. DT exhibited higher variability of peak frequency and greater instability of tremor burst intervals over time (higher tremor stability index) than ET or TAWD regardless of tasks. Intermuscular coherence magnitude between the antagonist pairs increased during the writing task in DT, but not ET or TAWD. ET and TAWD exhibited different phase relationships of the temporal fluctuations of voluntary movement and tremor in the kinetic condition. A linear discriminant classifier based on these tremor parameters was able to distinguish the three groups with a classification accuracy of 95.1%. Cerebellar inhibition was significantly reduced in DT, but not in TAWD, compared with ET and healthy controls. Our study shows that the two DTS are distinct entities with DT closer to nontremorous dystonia and TAWD closer to ET.

SIGNIFICANCE STATEMENT This study provides novel findings about characteristics and pathophysiology of the two different types of dystonic tremor syndromes compared with essential tremor. Patients with DTS are classified into DT who have dystonia and tremor in the same area, and tremor associated with dystonia (TAWD) who have dystonia and tremor elsewhere. Our results showed that DT exhibits increased tremor variability, instability, and intermuscular coherence, and decreased cerebello-thalamo-cortical inhibition compared with TAWD. Our study shows that DT and TAWD are distinct phenotypes, and that the physiological characteristics of DT are more similar to nontremorous dystonia, and TAWD is closer to ET.

  • cerebellar function
  • dystonic tremor
  • essential tremor
  • pathophysiology
  • transcranial magnetic stimulation

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The Journal of Neuroscience: 40 (48)
Journal of Neuroscience
Vol. 40, Issue 48
25 Nov 2020
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The Pathophysiology of Dystonic Tremors and Comparison With Essential Tremor
Pattamon Panyakaew, Hyun Joo Cho, Sang Wook Lee, Tianxia Wu, Mark Hallett
Journal of Neuroscience 25 November 2020, 40 (48) 9317-9326; DOI: 10.1523/JNEUROSCI.1181-20.2020

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The Pathophysiology of Dystonic Tremors and Comparison With Essential Tremor
Pattamon Panyakaew, Hyun Joo Cho, Sang Wook Lee, Tianxia Wu, Mark Hallett
Journal of Neuroscience 25 November 2020, 40 (48) 9317-9326; DOI: 10.1523/JNEUROSCI.1181-20.2020
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Keywords

  • cerebellar function
  • dystonic tremor
  • essential tremor
  • pathophysiology
  • transcranial magnetic stimulation

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