The occurrence of dystonia in upper-limb multiple sclerosis tremor

Walt, Anneke van der; Buzzard, Katherine; Sung, Simon; Spelman, Tim; Kolbe, Scott; Marriott, Mark; Butzkueven, Helmut; Evans, Andrew

doi:10.1177/1352458515577690

Cited by 8 publications

(13 citation statements)

References 34 publications

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“…A movement disorder neurologist completed a clinical tremor assessment as has been previously described (Van der Walt et al 2015 ). Tremor in the affected limb was rated using the Bain score (Bain et al 1993 ).…”

Section: Methodsmentioning

confidence: 99%

“…The Bain score is a subjective rating scale from 0 to 10 (0 = no tremor and 10 = extremely severe) for overall tremor severity, tremor during handwriting and tremor during drawing of an Archimedes spiral on a pre-drawn pattern. Dystonia (Van der Walt et al 2015 ) in the tremor-affected arm was assessed using the Global Dystonia Scale (GDS) in the upper limb only (range 0–10).…”

Section: Methodsmentioning

confidence: 99%

“…In MS, cerebellar injury was traditionally thought to be the main pathological driver of MS tremor due to the predominant presence of intention tremor and the correlation between the severity of tremor and dysmetria, dysarthria and dysdiadochokinesia (Diener and Dichgans 1992 ). However, more recently, careful clinical evaluation of patients with MS tremor has highlighted the clinical commonalities with essential tremor and questioned the relevance of purely cerebellar features on clinical examination (Van der Walt et al 2012 , 2015 ). These two previous case studies observed the presence of lesions in the cerebellar peduncles (superior and middle) in patients with MS tremor as well as cerebellum (Fahn 1986 ; Nakamura et al 1993 ).…”

Section: Introductionmentioning

confidence: 99%

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Tremor in multiple sclerosis is associated with cerebello-thalamic pathology

et al. 2017

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Tremor in people with multiple sclerosis (MS) is a frequent and debilitating symptom with a relatively poorly understood pathophysiology. To determine the relationship between clinical tremor severity and structural magnetic resonance imaging parameters. Eleven patients with clinically definite MS and right-sided upper limb tremor were studied. Tremor severity was assessed using the Bain score (overall severity, writing, and Archimedes spiral drawing). Cerebellar dysfunction was assessed using the Scale for the Assessment and Rating of Ataxia. Dystonia was assessed using the Global Dystonia Scale adapted for upper limb. For all subjects, volume was calculated for the thalamus from T1-weighted volumetric scans using Freesurfer. Superior cerebellar peduncle (SCP) cross-sectional areas were measured manually. The presence of lesions was visually determined and the lesion volumes were calculated by the lesion growth algorithm as implemented in the Lesion Segmentation Toolbox. Right thalamic volume negatively correlated with Bain tremor severity score (ρ = − 0.65, p = 0.03). Left thalamic volume negatively correlated with general Bain tremor severity score (ρ = − 0.65, p = 0.03) and the Bain writing score (ρ = − 0.65, p = 0.03). Right SCP area negatively correlated with Bain writing score (ρ = − 0.69, p = 0.02). Finally, Bain Archimedes score was significantly higher in patients with lesions in the contralateral thalamus. Whole brain lesion load showed no relationship with tremor severity. These results implicate degeneration of key structures within the cerebello-thalamic pathway as pathological substrates for tremor in MS patients.

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Section: Methodsmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Tremor in multiple sclerosis is associated with cerebello-thalamic pathology

et al. 2017

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“…1 A combination of cerebellar dysfunction and dystonia underlies most multiple sclerosis upper limb tremors. 2 Similar mechanisms in our patient may contribute to PWT with injury within the cerebello-pallido-thalamo-cortical tract.…”

Section: Teaching Video Neuroimages: Primary Writing Tremormentioning

confidence: 86%

Teaching Video Neuro Images : Primary writing tremor

Kelly

Kelleher

O’Shea³

et al. 2016

Neurology

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A 30-year-old woman with recurring optic neuritis presented with tremor affecting her right hand when writing only (video at Neurology.org). An MRI brain (figure) revealed lesions in the left midbrain, right middle cerebellar peduncle, and left cerebellar hemisphere.Primary writing tremor (PWT) is a task-specific tremor that occurs with writing. There is debate about whether it represents essential tremor, dystonia associated with writer's cramp, or a separate entity. 1A combination of cerebellar dysfunction and dystonia underlies most multiple sclerosis upper limb tremors.2 Similar mechanisms in our patient may contribute to PWT with injury within the cerebello-pallido-thalamo-cortical tract. AUTHOR CONTRIBUTIONSDr. Kelly: acquisition of data, interpretation of data, drafting/revising the manuscript for content. Dr. Kelleher: drafting/revising the manuscript for content. Dr. O'Shea: interpretation of data, revising the manuscript for content. Dr. Sweeney: interpretation of data, drafting/revising the manuscript for content, study supervision. STUDY FUNDINGNo targeted funding reported. DISCLOSUREThe authors report no disclosures relevant to the manuscript. Go to Neurology.org for full disclosures.

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“…Tremor is one of the most common movement disorders and is a symptom of many neurological disorders including PD, ET, and MS ( 37 , 38 ). In MS, tremor often presents as an action tremor with a postural and intention tremor component ( 39 ). There have been no task fMRI studies in MS tremor patients and tasks used in fMRI studies of other neurological diseases causing upper limb dysfunction lack the complexity to elicit tremulous movement and ability to assess tremor in the whole upper limb.…”

Section: Discussionmentioning

confidence: 99%

Novel Functional MRI Task for Studying the Neural Correlates of Upper Limb Tremor

et al. 2018

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Introduction: Tremor of the upper limbs is a disabling symptom that is present during several neurological disorders and is currently without treatment. Functional MRI (fMRI) is an essential tool to investigate the pathophysiology of tremor and aid the development of treatment options. However, no adequately or standardized protocols for fMRI exists at present. Here we present a novel, online available fMRI task that could be used to assess the in vivo pathology of tremor.Objective: This study aims to validate the tremor-evoking potential of the fMRI task in a small group of tremor patients outside the scanner and assess the reproducibility of the fMRI task related activation in healthy controls.Methods: Twelve HCs were scanned at two time points (baseline and after 6-weeks). There were two runs of multi-band fMRI and the tasks included a “brick-breaker” joystick game. The game consisted of three conditions designed to control for most of the activation related to performing the task by contrasting the conditions: WATCH (look at the game without moving joystick), MOVE (rhythmic left/right movement of joystick without game), and PLAY (playing the game). Task fMRI was analyzed using FSL FEAT to determine clusters of activation during the different conditions. Maximum activation within the clusters was used to assess the ability to control for task related activation and reproducibility. Four tremor patients have been included to test ecological and construct validity of the joystick task by assessing tremor frequencies captured by the joystick.Results: In HCs the game activated areas corresponding to motor, attention and visual areas. Most areas of activation by our game showed moderate to good reproducibility (intraclass correlation coefficient (ICC) 0.531–0.906) with only inferior parietal lobe activation showing poor reproducibility (ICC 0.446). Furthermore, the joystick captured significantly more tremulous movement in tremor patients compared to HCs (p = 0.01) during PLAY, but not during MOVE.Conclusion: Validation of our novel task confirmed tremor-evoking potential and reproducibility analyses yielded acceptable results to continue further investigations into the pathophysiology of tremor. The use of this technique in studies with tremor patient will no doubt provide significant insights into the treatment options.

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The occurrence of dystonia in upper-limb multiple sclerosis tremor

Abstract: Upper limb dystonia is common in MS tremor suggesting that MS tremor pathophysiology involves cerebello-pallido-thalamo-cortical network dysfunction.

Cited by 8 publications

References 34 publications

Tremor in multiple sclerosis is associated with cerebello-thalamic pathology

Tremor in multiple sclerosis is associated with cerebello-thalamic pathology

Teaching Video Neuro Images : Primary writing tremor

Novel Functional MRI Task for Studying the Neural Correlates of Upper Limb Tremor

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