TY - JOUR
T1 - Thalamic volume is reduced in cervical and laryngeal dystonias
AU - Waugh, Jeff L.
AU - Kuster, John K.
AU - Levenstein, Jacob M.
AU - Makris, Nikos
AU - Multhaupt-Buell, Trisha J.
AU - Sudarsky, Lewis R.
AU - Breiter, Hans C.
AU - Sharma, Nutan
AU - Blood, Anne J.
N1 - Funding Information:
Dr. Waugh was supported by the American Academy of Neurology's Clinical Research Training Fellowship and the Silverman Family Fellowship, Bachmann-Strauss Dystonia Parkinson Foundation. This work was supported by grants from the National Institute of Neurological Disorders and Stroke (grant number R01NS052368 to Dr. Blood, and grant number NINP50NS037409 to Dr. Breakefield with Dr. Sharma as PI of the clinical core), and a grant from the Dystonia Medical Research Foundation to Dr. Blood. Drs. Breiter, Blood, and Makris were funded by the National Institute on Drug Abuse (grant number R01 027804). Further support to Drs. Breiter and Blood came from grants (DABK39-03-0098 and DABK39-03-C-0098; The Phenotype Genotype Project in Addiction and Mood Disorder) from the Office of National Drug Control Policy - Counterdrug Technology Assessment Center, Washington, D.C. Further support was provided to Dr. Breiter by the Warren Wright Adolescent Center at Northwestern Memorial Hospital and Northwestern University, Chicago Il. The general infrastructure of the Martinos Center for Biomedical Imaging in which research on these grants was conducted, was supported by National Center for Research Resources (grant number P41 RR14075 to Dr. Rosen), and the Mental Illness and Neuroscience Discovery (MIND) Institute (Dr. Rosen). Shared processing cluster and secure storage was provided by Shared Instrumentation Grants 1S10RR023043 and 1S10RR023401. Prescreening and exams were conducted in the General Clinical Research Center, funded by National Center for Research Resources (grant number UL1 RR025758-01). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.
PY - 2016/5/1
Y1 - 2016/5/1
N2 - 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.
AB - 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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U2 - 10.1371/journal.pone.0155302
DO - 10.1371/journal.pone.0155302
M3 - Article
C2 - 27171035
AN - SCOPUS:84969534982
VL - 11
JO - PLoS One
JF - PLoS One
SN - 1932-6203
IS - 5
M1 - e0155302
ER -