Microstructural Abnormalities of the Dentatorubrothalamic Tract in Cervical Dystonia

Sondergaard, Rachel; Rockel, Conrad; Cortese, Filomeno; Jasaui, Yamile; Pringsheim, Tamara; Sarna, Justyna R.; Monchi, Oury; Sadikot, Abbas F.; Pike, Bruce; Martino, Davide

doi:10.1002/mds.28649

Cited by 15 publications

(31 citation statements)

References 46 publications

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“…12,13 Last, we found no significant correlation between FC maps and clinical scores. This finding is consistent with previous studies on focal dystonia that did not disclose significant correlations between clinical features and imaging parameters 37 and may occur when tests are partly self-administered.…”

Section: Discussionsupporting

confidence: 93%

“…Consistently, FC normalization of the thalamus and putamen with the sensorimotor cortex after botulinum toxin treatment was reported by Brodoehl et al 4 Dentate FC alterations in CD may also represent a functional correlate of microstructural changes characterized by decreased axonal integrity in the dentato-rubro-thalamic tract, which is known to be an important relay between sensorimotor network areas. 37 We may speculate that increased FC between the dentate nucleus and thalamus is a mechanism to compensate for structural degeneration. In line with recent literature, 37 our results support evidence of cerebellar outflow pathway alterations in CD.…”

Section: Discussionmentioning

confidence: 98%

“…37 We may speculate that increased FC between the dentate nucleus and thalamus is a mechanism to compensate for structural degeneration. In line with recent literature, 37 our results support evidence of cerebellar outflow pathway alterations in CD.…”

Section: Discussionmentioning

confidence: 98%

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Focal Dystonia

et al. 2022

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Background and ObjectivesNeuroimaging studies suggest that changes in the cerebellar-basal ganglia-thalamo-cortical sensorimotor circuit are a pathophysiologic feature of focal dystonia. However, it remains unclear whether structural and functional alterations vary in different forms of focal dystonia. Thus, in patients with cervical dystonia (CD) and blepharospasm (BSP), we aimed to investigate structural damage and resting-state functional alterations using whole-brain and seed-based approaches to test the hypothesis of possible functional connectivity (FC) alterations in specific circuits, including the cerebellum, basal ganglia, and cerebral cortex, in the context of preserved global FC.MethodsIn this cross-sectional study, we applied a multimodal 3T MRI protocol, including 3-dimensional T1-weighted images to extract brain volumes and cortical thickness, and fMRI at rest to study FC of the dentate nucleus and globus pallidus with a seed-based approach and whole-brain FC with a graph theory approach.ResultsThis study included 33 patients (17 with CD [14 female] age 55.7 ± 10.1 years, 16 with BSP [11 female] age 62.9 ± 8.8 years) and 16 age- and sex-matched healthy controls (HC) (7 female) 54.3 ± 14.3 years if age. Patients with CD, patients with BSP, and HC did not differ in terms of cortical or subcortical volume. Compared to HC, both patients with CD and patients with BSP had a loss of dentate FC anticorrelation with the sensorimotor cortex. Patients with CD and those with BSP showed increased pallidal FC with the cerebellum, supplementary motor area, and prefrontal cortices with respect to HC. Increased dentate FC with the cerebellum and thalamus and increased pallidal FC with the bilateral thalamus, sensorimotor and temporo-occipital cortices, and right putamen were present in patients with CD but not patients with BSP compared to HC. Measures of global FC, that is, global efficiency and small-worldness, did not differ between patients and HC.DiscussionBoth patients with CD and those with BSP showed altered dentate and pallidal FC with regions belonging to the integrated cerebellar-basal ganglia-thalamo-cortical sensorimotor circuit, supporting the concept that focal dystonia is a disorder of specific networks and not merely a result of basal ganglia alterations in the context of a preserved whole-brain functional architecture. Differences in functional interplay among specific brain structures may distinguish CD and BSP.

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Section: Discussionsupporting

confidence: 93%

Section: Discussionmentioning

confidence: 98%

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Focal Dystonia

et al. 2022

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“…Two very recent cases describe similar improvement in tonic components of craniocervical dystonia after bilateral Vim DBS [33,34]. It is thus possible that in some patients with dystonia, the underlying "circuitopathy" is localized to the cerebellothalamic pathways, as has been suggested by functional and structural neuroimaging [35][36][37]. In these patients, a shared cerebellar pathophysiology may produce both dystonia and an ET-like upper limb postural and action tremor, which would then both respond to DBS of the cerebello-thalamic relay in the Vim.…”

Section: Discussionmentioning

confidence: 88%

Vim-Thalamic Deep Brain Stimulation for Cervical Dystonia and Upper-Limb Tremor: Quantification by Markerless-3D Kinematics and Accelerometry

Mason

Cross

Arac

et al. 2022

Tremor and Other Hyperkinetic Movements

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Background: Deep Brain Stimulation (DBS) for dystonia is usually targeted to the globus pallidus internus (GPi), though stimulation of the ventral-intermediate nucleus of the thalamus (Vim) can be an effective treatment for phasic components of dystonia including tremor. We report on a patient who developed a syndrome of bilateral upper limb postural and action tremor and progressive cervical dystonia with both phasic and tonic components which were responsive to Vim DBS. We characterize and quantify this effect using markerless-3D-kinematics combined with accelerometry.Methods: Stereo videography was used to record our subject in 3D. The DeepBehavior toolbox was applied to obtain timeseries of joint position for kinematic analysis [1]. Accelerometry was performed simultaneously for comparison with prior literature.Results: Bilateral Vim DBS improved both dystonic tremor magnitude and tonic posturing. DBS of the hemisphere contralateral to the direction of dystonic head rotation (left Vim) had greater efficacy. Assessment of tremor magnitude by 3D-kinematics was concordant with accelerometry and was able to quantify tonic dystonic posturing.Discussion: In this case, Vim DBS treated both cervical dystonic tremor and dystonic posturing. Markerless-3D-kinematics should be further studied as a method of quantifying and characterizing tremor and dystonia.

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“…Of the studies identified, the number of diffusion directions ranged from 6 ( n = 5) [9–13] to ≥60 ( n = 18) [14, 31, 33, 35, 37, 39, 42–45, 49–51, 54, 57–59, 63], the remaining studies using an intermediate number of directions ( n = 10) [30, 32, 34, 36, 38, 40, 41, 46, 52, 53, 55, 56, 60–62, 64–68, 70] or no details were provided ( n = 1) [15]. The b values employed were in typical ranges of 700–1000 s/mm 2 , with five higher at 1100–1500 s/mm 2 [38, 39, 49–51].…”

Section: Resultsmentioning

confidence: 99%

Structural magnetic resonance imaging in dystonia: A systematic review of methodological approaches and findings

MacIver

Tax

Jones

et al. 2022

Euro J of Neurology

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Background and purpose: Structural magnetic resonance techniques have been widely applied in neurological disorders to better understand tissue changes, probing characteristics such as volume, iron deposition and diffusion. Dystonia is a hyperkinetic movement disorder, resulting in abnormal postures and pain. Its pathophysiology is poorly understood, with normal routine clinical imaging in idiopathic forms. More advanced tools provide an opportunity to identify smaller scale structural changes which may underpin pathophysiology. This review aims to provide an overview of methodological approaches undertaken in structural brain imaging of dystonia cohorts, and to identify commonly identified pathways, networks or regions that are implicated in pathogenesis.Methods: Structural magnetic resonance imaging studies of idiopathic and genetic forms of dystonia were systematically reviewed. Adhering to strict inclusion and exclusion criteria, PubMed and Embase databases were searched up to January 2022, with studies reviewed for methodological quality and key findings. Results:Seventy-seven studies were included, involving 1945 participants. The majority of studies employed diffusion tensor imaging (DTI) (n = 45) or volumetric analyses (n = 37), with frequently implicated areas of abnormality in the brainstem, cerebellum, basal ganglia and sensorimotor cortex and their interconnecting white matter pathways. Genotypic and motor phenotypic variation emerged, for example fewer cerebellothalamic tractography streamlines in genetic forms than idiopathic and higher grey matter volumes in task-specific than non-task-specific dystonias.Discussion: Work to date suggests microstructural brain changes in those diagnosed with dystonia, although the underlying nature of these changes remains undetermined.Employment of techniques such as multiple diffusion weightings or multi-exponential relaxometry has the potential to enhance understanding of these differences.

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Microstructural Abnormalities of the Dentatorubrothalamic Tract in Cervical Dystonia

Cited by 15 publications

References 46 publications

Focal Dystonia

Focal Dystonia

Vim-Thalamic Deep Brain Stimulation for Cervical Dystonia and Upper-Limb Tremor: Quantification by Markerless-3D Kinematics and Accelerometry

Structural magnetic resonance imaging in dystonia: A systematic review of methodological approaches and findings

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