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October 12, 2020Article

Dystonia & tremor: A cross-sectional study of the dystonia coalition cohort

Aasef G. Shaikh, View ORCID ProfileSinem Balta Beylergil, Laura Scorr, Gamze Kilic-Berkmen, Alan Freeman, Christine Klein, Johanna Junker, Sebastian Loens, Norbert Brüggemann, Alexander Münchau, Tobias Bäumer, Marie Vidailhet, Emmanuel Roze, Cecilia Bonnet, Joseph Jankovic, Joohi Jimenez-Shahed, Neepa Patel, Laura Marsh, Cynthia Comella, Richard L. Barbano, View ORCID ProfileBrian D. Berman, Irene Malaty, Aparna Wagle Shukla, Stephen G. Reich, Mark S. Ledoux, Alfredo Berardelli, Gina Ferrazzano, Natividad Stover, William Ondo, Sarah Pirio Richardson, Rachel Saunders-Pullman, Zoltan Mari, Pinky Agarwal, Charles Adler, Sylvain Chouinard, Susan H. Fox, View ORCID ProfileAllison Brashear, Daniel Truong, Oksana Suchowersky, Samuel Frank, Stewart Factor, Joel Perlmutter, H. A. Jinnah
First published October 12, 2020, DOI: https://doi.org/10.1212/WNL.0000000000011049
Aasef G. Shaikh
1Department of Biomedical Engineering, Case Western University School of Medicine, Cleveland, OH, USA; Department of Neurology, Case Western University School of Medicine, Cleveland, OH, USA; Neurological Institute, University Hospitals, Cleveland, OH, USA; Neurology Service, Louis Stokes Cleveland VA Medical Center, Cleveland, OH, USA.
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Sinem Balta Beylergil
2Department of Biomedical Engineering, Case Western University School of Medicine, Cleveland, OH, USA.
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Laura Scorr
3Department of Neurology, Emory University School of Medicine, Atlanta, Georgia, USA.
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Gamze Kilic-Berkmen
3Department of Neurology, Emory University School of Medicine, Atlanta, Georgia, USA.
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Alan Freeman
3Department of Neurology, Emory University School of Medicine, Atlanta, Georgia, USA.
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Christine Klein
4Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
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Johanna Junker
4Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
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Sebastian Loens
4Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
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Norbert Brüggemann
4Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
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Alexander Münchau
4Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
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Tobias Bäumer
4Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
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Marie Vidailhet
5Department of Neurology, Pitié-Salpêtrière Hospital, Paris, France.
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Emmanuel Roze
5Department of Neurology, Pitié-Salpêtrière Hospital, Paris, France.
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Cecilia Bonnet
5Department of Neurology, Pitié-Salpêtrière Hospital, Paris, France.
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Joseph Jankovic
6Department of Neurology, Baylor College of Medicine, Houston, TX, USA.
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Joohi Jimenez-Shahed
7Neurology and Neurosurgery, Icahn School of Medicine at Mount Sinai, New York, NY, USA.
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Neepa Patel
8Department of Neurology, Henry Ford Health System, West Bloomfield, Michigan, USA.
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Laura Marsh
9Department of Psychiatry and Neurology, Baylor College of Medicine, Houston, TX, USA.
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Cynthia Comella
10Department of Neurological Sciences, Rush University Medical Center, Chicago, IL, USA.
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Richard L. Barbano
11Department of Neurology, University of Rochester, Rochester, New York, USA.
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Brian D. Berman
12Department of Neurology, University of Colorado School of Medicine, Aurora, CO, United States.
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Irene Malaty
13Department of Neurology, University of Florida, Norman Fixel Institute for Neurological Diseases, Gainesville, FL, USA.
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Aparna Wagle Shukla
13Department of Neurology, University of Florida, Norman Fixel Institute for Neurological Diseases, Gainesville, FL, USA.
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Stephen G. Reich
14Department of Neurology, University of Maryland School of Medicine, Baltimore, MD USA.
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Mark S. Ledoux
15University of Tennessee Health Science Center, Memphis, Tennessee, USA.
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Alfredo Berardelli
16Department of Neurosciences, Mental Health and Sensory Organs, Suicide Prevention Center, Sant'Andrea Hospital, Sapienza University of Rome, Rome, Italy.
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Gina Ferrazzano
17IRCCS Neuromed, Via Atinense 18, 86077, Pozzilli, IS, Italy.
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Natividad Stover
18The University of Alabama at Birmingham, Birmingham, AL.
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William Ondo
19Methodist Neurological Institute, Houston, TX, USA.
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Sarah Pirio Richardson
20Department of Neurology, University of New Mexico Health Sciences Center, Albuquerque, NM, USA.
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Rachel Saunders-Pullman
21Department of Neurology, Mount Sinai Beth Israel, New York, New York.
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Zoltan Mari
22Lou Ruvo Center for Brain Health, Cleveland Clinic, Las Vegas, NV
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Pinky Agarwal
23Booth Gardner Parkinson's Care Center, Kirkland, Washington, USA.
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Charles Adler
24Mayo Clinic, Scottsdale, AZ, USA.
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Sylvain Chouinard
25Andre Barbeau Movement Disorders Unit, Montreal University Hospital Center (CHUM), QC, Canada.
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Susan H. Fox
26Movement Disorder Clinic, Toronto Western Hospital, Division of Neurology University of Toronto, Toronto, Ontario, Canada.
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Allison Brashear
27Dean, UC Davis School of Medicine, Sacramento, CA.
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Daniel Truong
28The Parkinson's and Movement Disorder Institute, Orange Coast Memorial Medical Center, Fountain Valley, CA.
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Oksana Suchowersky
29Department of Medicine, Medical Genetics, and Pediatrics, University of Alberta, Alberta, Canada.
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Samuel Frank
30Department of Neurology, Beth Israel Deaconess Medical Center, Boston, Massachusetts, USA.
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Stewart Factor
3Department of Neurology, Emory University School of Medicine, Atlanta, Georgia, USA.
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Joel Perlmutter
31Neurology, Radiology, Neuroscience, Physical Therapy and Occupational Therapy, Washington University School of Medicine, St Louis, Missouri.
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H. A. Jinnah
32Departments of Neurology, Human Genetics and Pediatrics, Emory University, Atlanta, GA, USA.
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Full PDF
Citation
Dystonia & tremor: A cross-sectional study of the dystonia coalition cohort
Aasef G. Shaikh, Sinem Balta Beylergil, Laura Scorr, Gamze Kilic-Berkmen, Alan Freeman, Christine Klein, Johanna Junker, Sebastian Loens, Norbert Brüggemann, Alexander Münchau, Tobias Bäumer, Marie Vidailhet, Emmanuel Roze, Cecilia Bonnet, Joseph Jankovic, Joohi Jimenez-Shahed, Neepa Patel, Laura Marsh, Cynthia Comella, Richard L. Barbano, Brian D. Berman, Irene Malaty, Aparna Wagle Shukla, Stephen G. Reich, Mark S. Ledoux, Alfredo Berardelli, Gina Ferrazzano, Natividad Stover, William Ondo, Sarah Pirio Richardson, Rachel Saunders-Pullman, Zoltan Mari, Pinky Agarwal, Charles Adler, Sylvain Chouinard, Susan H. Fox, Allison Brashear, Daniel Truong, Oksana Suchowersky, Samuel Frank, Stewart Factor, Joel Perlmutter, H. A. Jinnah
Neurology Oct 2020, 10.1212/WNL.0000000000011049; DOI: 10.1212/WNL.0000000000011049

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Abstract

Objective: To assess the clinical manifestations and predictors of different types of tremors in a individuals with different types of isolated dystonia.

Methods: Clinical manifestations of tremor were assessed in a multicenter, international cross-sectional, cohort study of 2362 individuals with all types of isolated dystonia (focal, segmental, multifocal and generalized) recruited through the Dystonia Coalition.

Results: Methodical and standardized assessments of all subjects in this cohort revealed the overall prevalence of any type of tremor was 53.3%. The prevalence of dystonic tremor varied from 36.9-48.4%, 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 two 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 eight subgroups within the whole cohort; defined largely by body region affected 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.

  • Received March 10, 2020.
  • Accepted in final form September 11, 2020.
  • © 2020 American Academy of Neurology

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