Aasef G Shaikh1, Sinem Balta Beylergil1, Laura Scorr1, Gamze Kilic-Berkmen1, Alan Freeman1, Christine Klein1, Johanna Junker1, Sebastian Loens1, Norbert Brüggemann1, Alexander Münchau1, Tobias Bäumer1, Marie Vidailhet1, Emmanuel Roze1, Cecilia Bonnet1, Joseph Jankovic1, Joohi Jimenez-Shahed1, Neepa Patel1, Laura Marsh1, Cynthia Comella1, Richard L Barbano1, Brian D Berman1, Irene Malaty1, Aparna Wagle Shukla1, Stephen G Reich1, Mark S Ledoux1, Alfredo Berardelli1, Gina Ferrazzano1, Natividad Stover1, William Ondo1, Sarah Pirio Richardson1, Rachel Saunders-Pullman1, Zoltan Mari1, Pinky Agarwal1, Charles Adler1, Sylvain Chouinard1, Susan H Fox1, Allison Brashear1, Daniel Truong1, Oksana Suchowersky1, Samuel Frank1, Stewart Factor1, Joel Perlmutter1, Hyder Azad Jinnah2. 1. 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. 2. 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. hjinnah@emory.edu.
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.
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.
Authors: Vikram G Shakkottai; Amit Batla; Kailash Bhatia; William T Dauer; Christian Dresel; Martin Niethammer; David Eidelberg; Robert S Raike; Yoland Smith; H A Jinnah; Ellen J Hess; Sabine Meunier; Mark Hallett; Rachel Fremont; Kamran Khodakhah; Mark S LeDoux; Traian Popa; Cécile Gallea; Stéphane Lehericy; Andreea C Bostan; Peter L Strick Journal: Cerebellum Date: 2017-04 Impact factor: 3.847
Authors: Dietrich Haubenberger; Giovanni Abbruzzese; Peter G Bain; Nin Bajaj; Julián Benito-León; Kailash P Bhatia; Günther Deuschl; Maria João Forjaz; Mark Hallett; Elan D Louis; Kelly E Lyons; Tiago A Mestre; Jan Raethjen; Maria Stamelou; Eng-King Tan; Claudia M Testa; Rodger J Elble Journal: Mov Disord Date: 2016-06-06 Impact factor: 10.338
Authors: Kailash P Bhatia; Peter Bain; Nin Bajaj; Rodger J Elble; Mark Hallett; Elan D Louis; Jan Raethjen; Maria Stamelou; Claudia M Testa; Guenther Deuschl Journal: Mov Disord Date: 2017-11-30 Impact factor: 10.338
Authors: Maria Stamelou; Gavin Charlesworth; Carla Cordivari; Susanne A Schneider; Georg Kägi; Una-Marie Sheerin; Ignacio Rubio-Agusti; Amit Batla; Henry Houlden; Nicholas W Wood; Kailash P Bhatia Journal: Mov Disord Date: 2014-01-17 Impact factor: 10.338
Authors: Gamze Kilic-Berkmen; Sarah Pirio Richardson; Joel S Perlmutter; Mark Hallett; Christine Klein; Aparna Wagle-Shukla; Irene A Malaty; Stephen G Reich; Brian D Berman; Jeanne Feuerstein; Marie Vidailhet; Emmanuel Roze; Joseph Jankovic; Abhimanyu Mahajan; Alberto J Espay; Richard L Barbano; Mark S LeDoux; Alexander Pantelyat; Samuel Frank; Natividad Stover; Alfredo Berardelli; Julie Leegwater-Kim; Giovanni Defazio; Scott A Norris; Hyder A Jinnah Journal: Mov Disord Clin Pract Date: 2021-12-01
Authors: Maggie Fagan; Laura Scorr; Doug Bernhardt; Ellen J Hess; Joel S Perlmutter; Carlos A Pardo; H A Jinnah Journal: Exp Neurol Date: 2021-08-28 Impact factor: 5.330
Authors: Johanna Junker; Brian D Berman; James Hall; Deena W Wahba; Valerie Brandt; Joel S Perlmutter; Joseph Jankovic; Irene A Malaty; Aparna Wagle Shukla; Stephen G Reich; Alberto J Espay; Kevin R Duque; Neepa Patel; Emmanuel Roze; Marie Vidailhet; H A Jinnah; Norbert Brüggemann Journal: J Neurol Neurosurg Psychiatry Date: 2021-02-09 Impact factor: 13.654
Authors: Jeanne P Vu; Elizabeth Cisneros; Ha Yeon Lee; Linh Le; Qiyu Chen; Xiaoyan A Guo; Ryin Rouzbehani; Joseph Jankovic; Stewart Factor; Christopher G Goetz; Richard L Barbano; Joel S Perlmutter; Hyder A Jinnah; Sarah Pirio Richardson; Glenn T Stebbins; Rodger Elble; Cynthia L Comella; David A Peterson Journal: J Neurol Sci Date: 2022-01-22 Impact factor: 4.553
Authors: Elizabeth Cisneros; Jeanne P Vu; Ha Yeon Lee; Qiyu Chen; Casey N Benadof; Zheng Zhang; Emily A Pettitt; Subhagya K Joshi; Richard L Barbano; Joseph Jankovic; Hyder A Jinnah; Joel S Perlmutter; Brian D Berman; Abhimanyu Mahajan; Christopher G Goetz; Glenn T Stebbins; Cynthia L Comella; David A Peterson Journal: Tremor Other Hyperkinet Mov (N Y) Date: 2021-06-23
Authors: Meng Wang; Tolulope Sajobi; Francesca Morgante; Charles Adler; Pinky Agarwal; Tobias Bäumer; Alfredo Berardelli; Brian D Berman; Joel Blumin; Max Borsche; Allison Brashear; Andres Deik; Kevin Duque; Alberto J Espay; Gina Ferrazzano; Jeanne Feuerstein; Susan Fox; Samuel Frank; Mark Hallett; Joseph Jankovic; Mark S LeDoux; Julie Leegwater-Kim; Abhimanyu Mahajan; Irene A Malaty; William Ondo; Alexander Pantelyat; Sarah Pirio-Richardson; Emmanuel Roze; Rachel Saunders-Pullman; Oksana Suchowersky; Daniel Truong; Marie Vidailhet; Aparna Wagle Shukla; Joel S Perlmutter; Hyder A Jinnah; Davide Martino Journal: Eur J Neurol Date: 2021-08-04 Impact factor: 6.288