期刊
PLOS ONE
卷 11, 期 5, 页码 -出版社
PUBLIC LIBRARY SCIENCE
DOI: 10.1371/journal.pone.0155302
关键词
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资金
- American Academy of Neurology's Clinical Research Training Fellowship
- Silverman Family Fellowship, Bachmann-Strauss Dystonia Parkinson Foundation
- National Institute of Neurological Disorders and Stroke [R01NS052368, NINP50NS037409]
- Dystonia Medical Research Foundation
- National Institute on Drug Abuse [R01 027804]
- Office of National Drug Control Policy - Counterdrug Technology Assessment Center, Washington, D.C. [DABK39-03-0098, DABK39-03-C-0098]
- Warren Wright Adolescent Center at Northwestern Memorial Hospital & Northwestern University, Chicago Il
- National Center for Research Resources [P41 RR14075, UL1 RR025758-01]
- Mental Illness and Neuroscience Discovery (MIND) Institute
- Shared Instrumentation Grants [1S10RR023043, 1S10RR023401]
Background Dystonia, a debilitating movement disorder characterized by abnormal fixed positions and/or twisting postures, is associated with dysfunction of motor control networks. While gross brain lesions can produce secondary dystonias, advanced neuroimaging techniques have been required to identify network abnormalities in primary dystonias. Prior neuroimaging studies have provided valuable insights into the pathophysiology of dystonia, but few directly assessed the gross volume of motor control regions, and to our knowledge, none identified abnormalities common to multiple types of idiopathic focal dystonia. Methods We used two gross volumetric segmentation techniques and one voxelwise volumetric technique (voxel based morphometry, VBM) to compare regional volume between matched healthy controls and patients with idiopathic primary focal dystonia (cervical, n = 17, laryngeal, n = 7). We used (1) automated gross volume measures of eight motor control regions using the FreeSurfer analysis package; (2) blinded, anatomist-supervised manual segmentation of the whole thalamus (also gross volume); and (3) voxel based morphometry, which measures local T1-weighted signal intensity and estimates gray matter density or volume at the level of single voxels, for both whole-brain and thalamus. Results Using both automated and manual gross volumetry, we found a significant volume decrease only in the thalamus in two focal dystonias. Decreases in whole-thalamic volume were independent of head and brain size, laterality of symptoms, and duration. VBM measures did not differ between dystonia and control groups in any motor control region. Conclusions Reduced thalamic gross volume, detected in two independent analyses, suggests a common anatomical abnormality in cervical dystonia and spasmodic dysphonia. Defining the structural underpinnings of dystonia may require such complementary approaches.
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