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January 26, 2021; 96 (4) Article

Dystonia and 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, Hyder Azad Jinnah
First published October 12, 2020, DOI: https://doi.org/10.1212/WNL.0000000000011049
Aasef G. Shaikh
From the Departments of Biomedical Engineering (A.G.S., S.B.B.) and Neurology (A.G.S.), Case Western University School of Medicine; Neurological Institute (A.G.S.), University Hospitals Cleveland; Neurology Service (A.G.S.), Louis Stokes Cleveland VA Medical Center, OH; Department of Neurology (L.S., G.K.-B., A.F., S. Factor, H.A.J.), Human Genetics (H.A.J.), and Pediatrics (H.A.J.), Emory University School of Medicine, Atlanta, GA; Institute of Neurogenetics (C.K., J.J., S.L., N.B., A.M., T.B.), University of Lübeck, Germany; Department of Neurology (M.V., E.R., C.B.), Pitié-Salpêtrière Hospital, Paris, France; Department of Neurology (J.J.), Baylor College of Medicine, Houston, TX; Neurology and Neurosurgery (J.J.-S.), Icahn School of Medicine at Mount Sinai, New York, NY; Department of Neurology (N.P.), Henry Ford Health System, West Bloomfield, MI; Department of Psychiatry and Neurology (L.M.), Baylor College of Medicine, Houston, TX; Department of Neurological Sciences (C.C.), Rush University Medical Center, Chicago, IL; Department of Neurology (R.L.B.), University of Rochester, NY; Department of Neurology (B.D.B.), University of Colorado School of Medicine, Aurora; Department of Neurology (I.M., A.W.S.), Norman Fixel Institute for Neurological Diseases, University of Florida, Gainesville; Department of Neurology (S.G.R.), University of Maryland School of Medicine, Baltimore; University of Tennessee Health Science Center (M.S.L.), Memphis; Department of Neurosciences (A.B.), Mental Health and Sensory Organs, Suicide Prevention Center, Sant'Andrea Hospital, Sapienza University of Rome; IRCCS Neuromed (G.F.), Pozzilli, Italy; The University of Alabama at Birmingham (N.S.); Methodist Neurological Institute (W.O.), Houston, TX; Department of Neurology (S.P.R.), University of New Mexico Health Sciences Center, Albuquerque; Department of Neurology (R.S.-P.), Mount Sinai Beth Israel, New York, NY; Lou Ruvo Center for Brain Health (Z.M.), Cleveland Clinic, Las Vegas, NV; Booth Gardner Parkinson's Care Center (P.A.), Kirkland, WA; Mayo Clinic (C.A.), Scottsdale, AZ; Andre Barbeau Movement Disorders Unit (S.C.), Montreal University Hospital Center (CHUM); Movement Disorder Clinic (S.H.F.), Toronto Western Hospital, Division of Neurology University of Toronto, Canada; UC Davis School of Medicine (A.B.), Sacramento; The Parkinson's and Movement Disorder Institute (D.T.), Orange Coast Memorial Medical Center, Fountain Valley, CA; Department of Medicine (O.S.), Medical Genetics, and Pediatrics, University of Alberta, Canada; Department of Neurology (S. Frank), Beth Israel Deaconess Medical Center, Boston, MA; and Neurology, Radiology, Neuroscience, Physical Therapy and Occupational Therapy (J.P.), Washington University School of Medicine, St Louis, MO.
MD, PhD
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Sinem Balta Beylergil
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Laura Scorr
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Gamze Kilic-Berkmen
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Alan Freeman
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Christine Klein
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Johanna Junker
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Sebastian Loens
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Norbert Brüggemann
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Alexander Münchau
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Tobias Bäumer
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Marie Vidailhet
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Emmanuel Roze
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Cecilia Bonnet
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Joseph Jankovic
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Joohi Jimenez-Shahed
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Neepa Patel
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Laura Marsh
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Cynthia Comella
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Richard L. Barbano
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Brian D. Berman
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Irene Malaty
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Aparna Wagle Shukla
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Stephen G. Reich
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Mark S. Ledoux
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Alfredo Berardelli
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Gina Ferrazzano
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Natividad Stover
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William Ondo
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Sarah Pirio Richardson
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Rachel Saunders-Pullman
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Zoltan Mari
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Pinky Agarwal
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Charles Adler
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Sylvain Chouinard
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Susan H. Fox
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Allison Brashear
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Daniel Truong
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Oksana Suchowersky
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Samuel Frank
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Stewart Factor
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Joel Perlmutter
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Hyder Azad Jinnah
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Citation
Dystonia and 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, Hyder Azad Jinnah
Neurology Jan 2021, 96 (4) e563-e574; DOI: 10.1212/WNL.0000000000011049

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Abstract

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.

Glossary

BFM=
Burke-Fahn-Marsden Dystonia Rating Scale;
GDRS=
Global Dystonia Rating Scale;
MDS=
Movement Disorders Society;
TAWD=
tremor associated with dystonia

Footnotes

  • Go to Neurology.org/N for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • CME Course: NPub.org/cmelist

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