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Predictors of alcohol responsiveness in dystonia

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.
0028-3878
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
58275f5c-3935-43c1-967e-c8f2a6596241
Weissbach, Anne
a068da20-adf2-48b4-8577-e5e242972a9e
Comella, Cynthia
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Malaty, Irene A.
1dbd03a1-fabe-4b91-a364-6edb5f6c5781
Jankovic, Joseph
084ab164-815e-450b-b049-35137c003201
LeDoux, Mark S.
d5591ba6-9360-4a5f-9a28-0613221b2a03
Berardelli, Alfredo
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Barbano, Richard
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Reich, Stephen G.
2a9621f0-8403-486e-9676-467a83f6a124
Perlmutter, Joel S.
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Jinnah, H.A.
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Brüggemann, Norbert
897a113b-3470-4f24-b434-11e838b94ab1
Junker, Johanna
fb0ba75f-9dfd-4045-ae04-ac2ad02b1a5c
Brandt, Valerie
e41f5832-70e4-407d-8a15-85b861761656
Berman, Brian M.
3366f65a-ca35-409a-904a-7b959d4f3fbb
Vidailhet, Marie
59d39a6d-0a7b-4525-8e0e-3eae0db744d3
Roze, Emmanuel
58275f5c-3935-43c1-967e-c8f2a6596241
Weissbach, Anne
a068da20-adf2-48b4-8577-e5e242972a9e
Comella, Cynthia
0bc17c32-9a4e-4c0b-9ed0-f7d1bddbb5d4
Malaty, Irene A.
1dbd03a1-fabe-4b91-a364-6edb5f6c5781
Jankovic, Joseph
084ab164-815e-450b-b049-35137c003201
LeDoux, Mark S.
d5591ba6-9360-4a5f-9a28-0613221b2a03
Berardelli, Alfredo
8ec0b805-1642-4758-b6a6-addab0a8527a
Barbano, Richard
1c3c579d-9af5-4b3a-8510-b46c41beed18
Reich, Stephen G.
2a9621f0-8403-486e-9676-467a83f6a124
Perlmutter, Joel S.
39120f4b-118e-453d-9fec-76875b5a3063
Jinnah, H.A.
2c1af19c-c05c-4853-8714-566958bfa6a8
Brüggemann, Norbert
897a113b-3470-4f24-b434-11e838b94ab1

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), E2020-E2026. (doi:10.1212/WNL.0000000000006551).

Record type: Article

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: Valerie Brandt
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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