TY - JOUR
T1 - Dystonia and Tremor: A Cross-Sectional Study of the Dystonia Coalition Cohort
AU - Shaikh, Aasef G.
AU - Beylergil, Sinem Balta
AU - Scorr, Laura
AU - Kilic-Berkmen, Gamze
AU - Freeman, Alan
AU - Klein, Christine
AU - Junker, Johanna
AU - Loens, Sebastian
AU - Brüggemann, Norbert
AU - Münchau, Alexander
AU - Bäumer, Tobias
AU - Vidailhet, Marie
AU - Roze, Emmanuel
AU - Bonnet, Cecilia
AU - Jankovic, Joseph
AU - Jimenez-Shahed, Joohi
AU - Patel, Neepa
AU - Marsh, Laura
AU - Comella, Cynthia
AU - Barbano, Richard L.
AU - Berman, Brian D.
AU - Malaty, Irene
AU - Wagle Shukla, Aparna
AU - Reich, Stephen G.
AU - Ledoux, Mark S.
AU - Berardelli, Alfredo
AU - Ferrazzano, Gina
AU - Stover, Natividad
AU - Ondo, William
AU - Pirio Richardson, Sarah
AU - Saunders-Pullman, Rachel
AU - Mari, Zoltan
AU - Agarwal, Pinky
AU - Adler, Charles
AU - Chouinard, Sylvain
AU - Fox, Susan H.
AU - Brashear, Allison
AU - Truong, Daniel
AU - Suchowersky, Oksana
AU - Frank, Samuel
AU - Factor, Stewart
AU - Perlmutter, Joel
AU - Jinnah, Hyder Azad
N1 - Publisher Copyright:
© 2020 American Academy of Neurology.
PY - 2021/1/26
Y1 - 2021/1/26
N2 - OBJECTIVE: To assess the clinical manifestations and predictors of different types of tremors in individuals with different types of isolated dystonia. METHODS: Clinical manifestations of tremor were assessed in a multicenter, international cross-sectional, cohort study of 2,362 individuals with all types of isolated dystonia (focal, segmental, multifocal, and generalized) recruited through the Dystonia Coalition. RESULTS: Methodical and standardized assessments of all participants in this cohort revealed the overall prevalence of any type of tremor was 53.3%. The prevalence of dystonic tremor varied from 36.9% to 48.4%, depending on criteria used to define it. To identify the factors associated with tremors in dystonia, the data were analyzed by generalized linear modeling and cluster analyses. Generalized linear modeling indicated 2 of the strongest factors associated with tremor included body region affected by dystonia and recruitment center. Tremor was also associated with severity of dystonia and duration of dystonia, but not with sex or race. The cluster analysis distinguished 8 subgroups within the whole cohort; defined largely by body region with dystonia, and secondarily by other clinical characteristics. CONCLUSION: The large number of cases evaluated by an international team of movement disorder experts facilitated the dissection of several important factors that influence the apparent prevalence and phenomenology of tremor in dystonia. These results are valuable for understanding the many differences reported in prior studies, and for guiding future studies of the nosology of tremor and dystonia.
AB - OBJECTIVE: To assess the clinical manifestations and predictors of different types of tremors in individuals with different types of isolated dystonia. METHODS: Clinical manifestations of tremor were assessed in a multicenter, international cross-sectional, cohort study of 2,362 individuals with all types of isolated dystonia (focal, segmental, multifocal, and generalized) recruited through the Dystonia Coalition. RESULTS: Methodical and standardized assessments of all participants in this cohort revealed the overall prevalence of any type of tremor was 53.3%. The prevalence of dystonic tremor varied from 36.9% to 48.4%, depending on criteria used to define it. To identify the factors associated with tremors in dystonia, the data were analyzed by generalized linear modeling and cluster analyses. Generalized linear modeling indicated 2 of the strongest factors associated with tremor included body region affected by dystonia and recruitment center. Tremor was also associated with severity of dystonia and duration of dystonia, but not with sex or race. The cluster analysis distinguished 8 subgroups within the whole cohort; defined largely by body region with dystonia, and secondarily by other clinical characteristics. CONCLUSION: The large number of cases evaluated by an international team of movement disorder experts facilitated the dissection of several important factors that influence the apparent prevalence and phenomenology of tremor in dystonia. These results are valuable for understanding the many differences reported in prior studies, and for guiding future studies of the nosology of tremor and dystonia.
UR - http://www.scopus.com/inward/record.url?scp=85100445475&partnerID=8YFLogxK
U2 - 10.1212/WNL.0000000000011049
DO - 10.1212/WNL.0000000000011049
M3 - Journal articles
C2 - 33046615
AN - SCOPUS:85100445475
SN - 0028-3878
VL - 96
SP - e563-e574
JO - Neurology
JF - Neurology
IS - 4
ER -