The Anatomical Basis for Dystonia: The Motor Network Model

Jinnah, H.A.; Neychev, Vladimir; Hess, Ellen J

doi:10.7916/d8v69x3s

Cited by 56 publications

(22 citation statements)

References 165 publications

(138 reference statements)

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“…3 Functional imaging studies have associated dystonia with abnormal activity across multiple motor and sensory brain regions, leading to the conception of a circuit disorder and supporting a network model. [4][5][6] This model is consistent with 3 main mechanisms believed to be linked to the physiopathology of dystonia: (a) disorders of the inhibitory process, (b) disorders of sensorimotor integration, 7 and (c) maladaptive plasticity. 3,8,9 From that perspective, several studies have shown abnormal cortical activity in patients with focal hand dystonia, suggesting a loss of surround inhibition 10,11 and altered somatosensory processing and integration.…”

Section: Introductionsupporting

confidence: 82%

Motor Cortex Activation During Writing in Focal Upper-Limb Dystonia: An fNIRS Study

Prôa

Balardin

Faria

et al. 2021

Neurorehabil Neural Repair

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Background Functional imaging studies have associated dystonia with abnormal activation in motor and sensory brain regions. Commonly used techniques such as functional magnetic resonance imaging impose physical constraints, limiting the experimental paradigms. Functional near-infrared spectroscopy (fNIRS) offers a new noninvasive possibility for investigating cortical areas and the neural correlates of complex motor behaviors in unconstrained settings. Methods We compared the cortical brain activation of patients with focal upper-limb dystonia and controls during the writing task under naturalistic conditions using fNIRS. The primary motor cortex (M1), the primary somatosensory cortex (S1), and the supplementary motor area were chosen as regions of interest (ROIs) to assess differences in changes in both oxyhemoglobin (oxy-Hb) and deoxyhemoglobin (deoxy-Hb) between groups. Results Group average activation maps revealed an expected pattern of contralateral recruitment of motor and somatosensory cortices in the control group and a more bilateral pattern of activation in the dystonia group. Between-group comparisons focused on specific ROIs revealed an increased activation of the contralateral M1 and S1 cortices and also of the ipsilateral M1 cortex in patients. Conclusions Overactivity of contralateral M1 and S1 in dystonia suggest a reduced specificity of the task-related cortical areas, whereas ipsilateral activation possibly indicates a primary disorder of the motor cortex or an endophenotypic pattern. To our knowledge, this is the first study using fNIRS to assess cortical activity in dystonia during the writing task under natural settings, outlining the potential of this technique for monitoring sensory and motor retraining in dystonia rehabilitation.

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Section: Introductionsupporting

confidence: 82%

Motor Cortex Activation During Writing in Focal Upper-Limb Dystonia: An fNIRS Study

Prôa

Balardin

Faria

et al. 2021

Neurorehabil Neural Repair

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“…This VBM study reviewed images of 73 patients with various forms of isolated idiopathic dystonia (focal, segmental, multifocal, and generalized dystonia), comprising one of the largest dystonia cohorts in a single-center till now. Results of our studies have revealed widespread GMV alterations involving somatosensory, motor, limbic and visual systems, supporting dystonia as a network disorder ( 34 ).…”

Section: Discussionsupporting

confidence: 62%

Imaging Insights of Isolated Idiopathic Dystonia: Voxel-Based Morphometry and Activation Likelihood Estimation Studies

Zhang

et al. 2022

Front. Neurol.

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The understanding of brain structural abnormalities across different clinical forms of dystonia and their contribution to clinical characteristics remains unclear. The objective of this study is to investigate shared and specific gray matter volume (GMV) abnormalities in various forms of isolated idiopathic dystonia. We collected imaging data from 73 isolated idiopathic dystonia patients and matched them with healthy controls to explore the GMV alterations in patients and their correlations with clinical characteristics using the voxel-based morphometry (VBM) technique. In addition, we conducted an activation likelihood estimation (ALE) meta-analysis of previous VBM studies. Our study demonstrated widespread morphometry alterations in patients with idiopathic dystonia. Multiple systems were affected, which mainly included basal ganglia, sensorimotor, executive control, and visual networks. As the result of the ALE meta-analysis, a convergent cluster with increased GMV was found in the left globus pallidus. In subgroup VBM analyses, decreased putamen GMV was observed in all clinic forms, while the increased GMV was observed in parahippocampal, lingual, and temporal gyrus. GD demonstrated the most extensive GMV abnormalities in cortical regions, and the aberrant GMV of the posterior cerebellar lobe was prominent in CD. Moreover, trends of increased GMV regions of the left precuneus and right superior frontal gyrus were demonstrated in the moderate-outcome group compared with the superior-outcome group. Results of our study indicated shared pathophysiology of the disease-centered on the dysfunction of the basal ganglia-thalamo-cortical circuit, impairing sensorimotor integration, high-level motor execution, and cognition of patients. Dysfunction of the cerebello-thalamo-cortical circuit could also be involved in CD especially. Finally, the frontal-parietal pathway may act as a potential marker for predicting treatment outcomes such as deep brain stimulation.

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“… 13 20 21 22 23 The Vo complex nucleus plays a key role in the motor function related with the basal ganglia-cerebello-thalamo-cortical circuit. 1 4 18 27 The pallidothalamic pathway involves both the GPi and Vo, and therefore, targeting both might provide more benefits. 28 Indeed, targeting the Vo complex has been found to elicit benefits in dystonic tremor or focal dystonia in previous studies and to sometimes show superiority over targeting only the GPi.…”

Section: Discussionmentioning

confidence: 99%

“… 3 In the past the pathophysiology of dystonia was thought to be the result of basal ganglia lesions; however, thanks to the development of many imaging and diagnostic techniques, recent studies suggest the disorder stems from dysfunction in the basal ganglia-cerebello-thalamo-cortical circuit. 4 …”

Section: Introductionmentioning

confidence: 99%

Dual Pallidal and Thalamic Deep Brain Stimulation for Complex Ipsilateral Dystonia

et al. 2022

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Purpose Globus pallidus pars interna (GPi) has become an established target for deep brain stimulation (DBS) in dystonia. Previous studies suggest that targeting the ventralis oralis (Vo) complex nucleus improves dystonic tremor or even focal dystonia. Research has also demonstrated that multi-target DBS shows some benefits over single target DBS. In this study, we reviewed patients who had undergone unilateral DBS targeting the GPi and Vo. Materials and Methods Five patients diagnosed with medically refractory upper extremity dystonia (focal or segmental) underwent DBS. Two DBS electrodes each were inserted unilaterally targeting the ipsilateral GPi and Vo. Clinical outcomes were evaluated using the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) and Disability Rating Scale. Results BFMDRS scores decreased by 55% at 1-month, 56% at 3-month, 59% at 6-month, and 64% at 12-month follow up. Disability Rating Scale scores decreased 41% at 1-month, 47% at 3-month, 50% at 6-month, and 60% at 12-month follow up. At 1 month after surgery, stimulating both targets improved clinical scores better than targeting GPi or Vo alone. Conclusion Unilateral thalamic and pallidal dual electrode DBS may be as effective or even superior to DBS of a single target for dystonia. Although the number of patients was small, our results reflected favorable clinical outcomes.

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The Anatomical Basis for Dystonia: The Motor Network Model

Cited by 56 publications

References 165 publications

Motor Cortex Activation During Writing in Focal Upper-Limb Dystonia: An fNIRS Study

Motor Cortex Activation During Writing in Focal Upper-Limb Dystonia: An fNIRS Study

Imaging Insights of Isolated Idiopathic Dystonia: Voxel-Based Morphometry and Activation Likelihood Estimation Studies

Dual Pallidal and Thalamic Deep Brain Stimulation for Complex Ipsilateral Dystonia

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