Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure
Objective: Isolated adult-onset focal dystonia is considered a network disorder with disturbances to the motor basal ganglia and cerebellar circuits playing a pathophysiological role, but why specific body regions become affected remains unknown. We aimed to use diffusion tensor imaging to determine...
| Main Authors: | , , , , |
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| Format: | Article |
| Language: | English |
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Elsevier
2018-01-01
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| Series: | NeuroImage: Clinical |
| Online Access: | http://www.sciencedirect.com/science/article/pii/S2213158218301876 |
| _version_ | 1818422914595160064 |
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| author | Brian D. Berman Justin M. Honce Erica Shelton Stefan H. Sillau Lidia M. Nagae |
| author_facet | Brian D. Berman Justin M. Honce Erica Shelton Stefan H. Sillau Lidia M. Nagae |
| author_sort | Brian D. Berman |
| collection | DOAJ |
| description | Objective: Isolated adult-onset focal dystonia is considered a network disorder with disturbances to the motor basal ganglia and cerebellar circuits playing a pathophysiological role, but why specific body regions become affected remains unknown. We aimed to use diffusion tensor imaging to determine if the two most common phenotypes of focal dystonia are associated with distinguishing microstructural changes affecting the motor network. Methods: Fifteen blepharospasm patients, 20 cervical dystonia patients, and 30 age- and sex-matched healthy controls were recruited. Maps of fractional anisotropy and mean diffusivity were analyzed using a voxel-based approach and an automated region-of-interest technique to evaluate deep gray matter nuclei. Correlations between diffusion measures and dystonia severity were tested, and post hoc discriminant analyses were conducted. Results: Voxel-based analyses revealed significantly reduced fractional anisotropy in the right cerebellum and increased mean diffusivity in the left caudate of cervical dystonia patients compared to controls, as well as lower fractional anisotropy in the right cerebellum in cervical dystonia patients relative to blepharospasm patients. In addition to reduced fractional anisotropy in the bilateral caudate nucleus of cervical dystonia patients relative to controls and blepharospasm patients, region-of-interest analyses revealed significantly reduced fractional anisotropy in the right globus pallidus internus and left red nucleus of blepharospasm patients compared to both controls and cervical dystonia patients. Diffusivity measures in the red nucleus of blepharospasm patients correlated with disease severity. In a three-group discriminant analysis, participants were correctly classified with only modest reliability (67–75%), but in a two-group discriminant analysis, patients could be distinguished from each other with high reliability (83–100%). Conclusions: Different focal dystonia phenotypes are associated with distinct patterns of altered microstructure within constituent regions of basal ganglia and cerebellar circuits. Keywords: Blepharospasm, Cervical dystonia, Diffusion tensor imaging, Basal ganglia, Cerebellum |
| first_indexed | 2024-12-14T13:33:49Z |
| format | Article |
| id | doaj.art-cf59f291e4d04a49a484e764f18ad6ff |
| institution | Directory Open Access Journal |
| issn | 2213-1582 |
| language | English |
| last_indexed | 2024-12-14T13:33:49Z |
| publishDate | 2018-01-01 |
| publisher | Elsevier |
| record_format | Article |
| series | NeuroImage: Clinical |
| spelling | doaj.art-cf59f291e4d04a49a484e764f18ad6ff2022-12-21T22:59:39ZengElsevierNeuroImage: Clinical2213-15822018-01-0119805812Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructureBrian D. Berman0Justin M. Honce1Erica Shelton2Stefan H. Sillau3Lidia M. Nagae4Department of Neurology, University of Colorado School of Medicine, Aurora, CO, United States; Department of Radiology, University of Colorado School of Medicine, Aurora, CO, United States; Neurology Section, Denver VA Medical Center, Denver, CO, United States; Corresponding author at: University of Colorado Anschutz Medical Campus, Department of Neurology, 12631 E. 17th Avenue, Mail Stop B-185, Aurora, CO 80045, United States.Department of Radiology, University of Colorado School of Medicine, Aurora, CO, United StatesDepartment of Radiology, University of Colorado School of Medicine, Aurora, CO, United StatesDepartment of Radiology, University of Colorado School of Medicine, Aurora, CO, United StatesDepartment of Radiology, University of Colorado School of Medicine, Aurora, CO, United States; Department of Radiology, University of Florida, Gainesville, FL, United StatesObjective: Isolated adult-onset focal dystonia is considered a network disorder with disturbances to the motor basal ganglia and cerebellar circuits playing a pathophysiological role, but why specific body regions become affected remains unknown. We aimed to use diffusion tensor imaging to determine if the two most common phenotypes of focal dystonia are associated with distinguishing microstructural changes affecting the motor network. Methods: Fifteen blepharospasm patients, 20 cervical dystonia patients, and 30 age- and sex-matched healthy controls were recruited. Maps of fractional anisotropy and mean diffusivity were analyzed using a voxel-based approach and an automated region-of-interest technique to evaluate deep gray matter nuclei. Correlations between diffusion measures and dystonia severity were tested, and post hoc discriminant analyses were conducted. Results: Voxel-based analyses revealed significantly reduced fractional anisotropy in the right cerebellum and increased mean diffusivity in the left caudate of cervical dystonia patients compared to controls, as well as lower fractional anisotropy in the right cerebellum in cervical dystonia patients relative to blepharospasm patients. In addition to reduced fractional anisotropy in the bilateral caudate nucleus of cervical dystonia patients relative to controls and blepharospasm patients, region-of-interest analyses revealed significantly reduced fractional anisotropy in the right globus pallidus internus and left red nucleus of blepharospasm patients compared to both controls and cervical dystonia patients. Diffusivity measures in the red nucleus of blepharospasm patients correlated with disease severity. In a three-group discriminant analysis, participants were correctly classified with only modest reliability (67–75%), but in a two-group discriminant analysis, patients could be distinguished from each other with high reliability (83–100%). Conclusions: Different focal dystonia phenotypes are associated with distinct patterns of altered microstructure within constituent regions of basal ganglia and cerebellar circuits. Keywords: Blepharospasm, Cervical dystonia, Diffusion tensor imaging, Basal ganglia, Cerebellumhttp://www.sciencedirect.com/science/article/pii/S2213158218301876 |
| spellingShingle | Brian D. Berman Justin M. Honce Erica Shelton Stefan H. Sillau Lidia M. Nagae Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure NeuroImage: Clinical |
| title | Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure |
| title_full | Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure |
| title_fullStr | Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure |
| title_full_unstemmed | Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure |
| title_short | Isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure |
| title_sort | isolated focal dystonia phenotypes are associated with distinct patterns of altered microstructure |
| url | http://www.sciencedirect.com/science/article/pii/S2213158218301876 |
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