Predictors of alcohol responsiveness in dystonia
Predictors of alcohol responsiveness in dystonia
Objective: To determine predictors of alcohol responsiveness in a large cohort of patients with dystonia.
Methods: A total of 2,159 participants with dystonia were prospectively enrolled in the cross-sectional Dystonia Coalition multicenter study. Patients with secondary, combined, or confirmed genetic dystonia (total n = 164) or unknown alcohol responsiveness (n = 737) were excluded. Patients answered a standardized questionnaire and were clinically examined using a standardized video protocol and the Burke-Fahn-Marsden Dystonia Rating Scale. Alcohol responsiveness was determined by patients' self-report.
Results: A total of 1,258 patients with isolated dystonia (mean age: 59.5 ± 12.2 years; 898 women) met the inclusion criteria; 369 patients (29.3%) reported improvement of dystonia after alcohol consumption. Alcohol responsiveness was not related to sex (p = 0.742), age (p = 0.715), or severity of dystonia (p = 0.623). Age at onset was lower in patients who responded to alcohol (p < 0.001). Alcohol responsiveness differed across dystonia subgroups (multifocal/generalized > segmental [p = 0.014]; cervical and laryngeal > cranial and limb [p < 0.001]) and was related to a positive family history of movement disorders (p = 0.001), and presence of tremor (p < 0.001).
Conclusion: The association of alcohol responsiveness with a positive family history for movement disorders, generalized dystonia, and an earlier age at onset suggests that patients with dystonia who have an underlying genetic contribution may be more likely to respond beneficially to alcohol. The fact that dystonic tremor may respond to alcohol is in keeping with the observation that the intake of GABAergic drugs may have a beneficial effect in a proportion of patients.
E2020-E2026
Junker, Johanna
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Brandt, Valerie
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Berman, Brian M.
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Vidailhet, Marie
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Roze, Emmanuel
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Weissbach, Anne
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Comella, Cynthia
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Malaty, Irene A.
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Jankovic, Joseph
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LeDoux, Mark S.
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Berardelli, Alfredo
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Barbano, Richard
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Reich, Stephen G.
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Perlmutter, Joel S.
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Jinnah, H.A.
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Brüggemann, Norbert
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20 November 2018
Junker, Johanna
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Brandt, Valerie
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Berman, Brian M.
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Vidailhet, Marie
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Roze, Emmanuel
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Weissbach, Anne
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Comella, Cynthia
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Malaty, Irene A.
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Jankovic, Joseph
084ab164-815e-450b-b049-35137c003201
LeDoux, Mark S.
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Berardelli, Alfredo
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Barbano, Richard
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Reich, Stephen G.
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Perlmutter, Joel S.
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Jinnah, H.A.
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Brüggemann, Norbert
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Junker, Johanna, Brandt, Valerie, Berman, Brian M., Vidailhet, Marie, Roze, Emmanuel, Weissbach, Anne, Comella, Cynthia, Malaty, Irene A., Jankovic, Joseph, LeDoux, Mark S., Berardelli, Alfredo, Barbano, Richard, Reich, Stephen G., Perlmutter, Joel S., Jinnah, H.A. and Brüggemann, Norbert
(2018)
Predictors of alcohol responsiveness in dystonia.
Neurology, 91 (21), .
(doi:10.1212/WNL.0000000000006551).
Abstract
Objective: To determine predictors of alcohol responsiveness in a large cohort of patients with dystonia.
Methods: A total of 2,159 participants with dystonia were prospectively enrolled in the cross-sectional Dystonia Coalition multicenter study. Patients with secondary, combined, or confirmed genetic dystonia (total n = 164) or unknown alcohol responsiveness (n = 737) were excluded. Patients answered a standardized questionnaire and were clinically examined using a standardized video protocol and the Burke-Fahn-Marsden Dystonia Rating Scale. Alcohol responsiveness was determined by patients' self-report.
Results: A total of 1,258 patients with isolated dystonia (mean age: 59.5 ± 12.2 years; 898 women) met the inclusion criteria; 369 patients (29.3%) reported improvement of dystonia after alcohol consumption. Alcohol responsiveness was not related to sex (p = 0.742), age (p = 0.715), or severity of dystonia (p = 0.623). Age at onset was lower in patients who responded to alcohol (p < 0.001). Alcohol responsiveness differed across dystonia subgroups (multifocal/generalized > segmental [p = 0.014]; cervical and laryngeal > cranial and limb [p < 0.001]) and was related to a positive family history of movement disorders (p = 0.001), and presence of tremor (p < 0.001).
Conclusion: The association of alcohol responsiveness with a positive family history for movement disorders, generalized dystonia, and an earlier age at onset suggests that patients with dystonia who have an underlying genetic contribution may be more likely to respond beneficially to alcohol. The fact that dystonic tremor may respond to alcohol is in keeping with the observation that the intake of GABAergic drugs may have a beneficial effect in a proportion of patients.
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Accepted/In Press date: 9 August 2018
e-pub ahead of print date: 19 October 2018
Published date: 20 November 2018
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Local EPrints ID: 424451
URI: http://eprints.soton.ac.uk/id/eprint/424451
ISSN: 0028-3878
PURE UUID: 3c04cbad-7672-4278-a202-1c604ffe993e
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Date deposited: 05 Oct 2018 11:37
Last modified: 15 Mar 2024 21:27
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Contributors
Author:
Johanna Junker
Author:
Brian M. Berman
Author:
Marie Vidailhet
Author:
Emmanuel Roze
Author:
Anne Weissbach
Author:
Cynthia Comella
Author:
Irene A. Malaty
Author:
Joseph Jankovic
Author:
Mark S. LeDoux
Author:
Alfredo Berardelli
Author:
Richard Barbano
Author:
Stephen G. Reich
Author:
Joel S. Perlmutter
Author:
H.A. Jinnah
Author:
Norbert Brüggemann
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