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

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 id...

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Published inFrontiers in neurology Vol. 13; p. 823882
Main Authors Wu, Yunhao, Zhang, Chao, Li, Yufei, Feng, Jie, Zhang, Ming, Li, Hongxia, Wang, Tao, Zhang, Yingying, Jin, Zhijia, Zhang, Chencheng, Zhang, Yuyao, Li, Dianyou, Wu, Yiwen, Wei, Hongjiang, Sun, Bomin
Format Journal Article
LanguageEnglish
Published Switzerland Frontiers Media S.A 26.04.2022
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Summary: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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Edited by: Letizia Leocani, San Raffaele Hospital (IRCCS), Italy
Reviewed by: Antonio Suppa, Sapienza University of Rome, Italy; Joshua K. Wong, University of Florida, United States
This article was submitted to Movement Disorders, a section of the journal Frontiers in Neurology
These authors have contributed equally to this work and share first authorship
These authors have contributed equally to this work and share last authorship
ISSN:1664-2295
1664-2295
DOI:10.3389/fneur.2022.823882