The University of Southampton
University of Southampton Institutional Repository

Quality of life in isolated dystonia: Non-motor manifestations matter

Quality of life in isolated dystonia: Non-motor manifestations matter
Quality of life in isolated dystonia: Non-motor manifestations matter
Objective: To evaluate the relationship between health-related quality of life (HR-QoL) and both physical and psychiatric factors in a large, international, multicenter cohort of isolated dystonia patients, the Dystonia Coalition.
Methods: Natural history data from 603 patients with isolated dystonia (median age 57 years (IQR: 48-64 years), 67.0% female) were prospectively acquired and analyzed. HR-QoL (RAND 36-Item Health Survey), severity of depressive symptoms, generalized anxiety (Hospital Anxiety and Depression Scale) and social anxiety (Liebowitz Social Anxiety Scale) were assessed. Dystonia severity (Burke-Fahn-Marsden Dystonia Rating Scale) and dystonic tremor were examined. Statistical predictors of HR-QoL were calculated using saturated path analysis.
Results: Reduced HR-QoL was strongly associated with the degree of depressive symptoms and generalized and social anxiety (8/8 RAND36 subscales, p ≤ .001). Increased dystonia severity was associated with worse physical functioning, physical and emotional role functioning, and social functioning (all p ≤ .001). The presence of tremor correlated with worse physical functioning and pain (all p ≤ .006). Younger age was associated with reduced emotional well-being and vitality (all p ≤ .006). There were no HR-QoL differences between sexes.
Conclusion: HR-QoL in isolated dystonia is strongly associated with psychiatric and physical features. While current standard of care focus on motor aspects of dystonia, comprehensive care should address both physical and mental aspects of health.
0022-3050
622-628
Junker, J.
2f29da6e-93c0-4544-bc18-2c320440d6a6
Berman, B.D.
271ee7e4-2f38-460e-bb41-c7003a99a513
Hall, J.
29e17a2b-dca0-4b91-be02-2ace4abaa6c4
Wahba, D.
dd795fb8-c72f-4064-97d1-1ed65d6a29ba
Brandt, V.
e41f5832-70e4-407d-8a15-85b861761656
Perlmutter, J.S.
01db8953-2976-4a06-b732-fb88d773bd65
Jankovic, J.
d7d7a530-ec7a-4f29-9a7a-c1410f21b983
Malaty, I.A.
1c15b4d1-46bc-41c6-84d7-15312caaf794
Shukla, A.W.
ed7f7065-43f6-4b94-bdc1-855e49f7e913
Reich, S.G.
daeba674-a7c4-4d12-93a0-3aaf7697c072
Espay, A.J.
2986da81-9f36-45f1-adbd-14c5e3249d18
Duque, K.R.
b4f40968-6449-410f-a0a3-6b401df35b93
Patel, N.
3d71c6a2-1b00-47a3-8480-607eb66a2cb3
Roze, E.
bd14c303-6c1f-4e14-b88d-2d1d08f96080
Vidailhet, M.
cef8fd23-412a-4c60-8a2f-3bbe0b4a9206
Jinnah, H.A.
f4ea4014-03ff-42a3-bc99-b7df13e4507e
Bruegemann, N.
7f61c882-5b16-4521-a450-295a2fa26736
Junker, J.
2f29da6e-93c0-4544-bc18-2c320440d6a6
Berman, B.D.
271ee7e4-2f38-460e-bb41-c7003a99a513
Hall, J.
29e17a2b-dca0-4b91-be02-2ace4abaa6c4
Wahba, D.
dd795fb8-c72f-4064-97d1-1ed65d6a29ba
Brandt, V.
e41f5832-70e4-407d-8a15-85b861761656
Perlmutter, J.S.
01db8953-2976-4a06-b732-fb88d773bd65
Jankovic, J.
d7d7a530-ec7a-4f29-9a7a-c1410f21b983
Malaty, I.A.
1c15b4d1-46bc-41c6-84d7-15312caaf794
Shukla, A.W.
ed7f7065-43f6-4b94-bdc1-855e49f7e913
Reich, S.G.
daeba674-a7c4-4d12-93a0-3aaf7697c072
Espay, A.J.
2986da81-9f36-45f1-adbd-14c5e3249d18
Duque, K.R.
b4f40968-6449-410f-a0a3-6b401df35b93
Patel, N.
3d71c6a2-1b00-47a3-8480-607eb66a2cb3
Roze, E.
bd14c303-6c1f-4e14-b88d-2d1d08f96080
Vidailhet, M.
cef8fd23-412a-4c60-8a2f-3bbe0b4a9206
Jinnah, H.A.
f4ea4014-03ff-42a3-bc99-b7df13e4507e
Bruegemann, N.
7f61c882-5b16-4521-a450-295a2fa26736

Junker, J., Berman, B.D., Hall, J., Wahba, D., Brandt, V., Perlmutter, J.S., Jankovic, J., Malaty, I.A., Shukla, A.W., Reich, S.G., Espay, A.J., Duque, K.R., Patel, N., Roze, E., Vidailhet, M., Jinnah, H.A. and Bruegemann, N. (2021) Quality of life in isolated dystonia: Non-motor manifestations matter. Journal of Neurology, Neurosurgery, and Psychiatry, 92 (6), 622-628, [jnnp-2020-325193]. (doi:10.1136/jnnp-2020-325193).

Record type: Article

Abstract

Objective: To evaluate the relationship between health-related quality of life (HR-QoL) and both physical and psychiatric factors in a large, international, multicenter cohort of isolated dystonia patients, the Dystonia Coalition.
Methods: Natural history data from 603 patients with isolated dystonia (median age 57 years (IQR: 48-64 years), 67.0% female) were prospectively acquired and analyzed. HR-QoL (RAND 36-Item Health Survey), severity of depressive symptoms, generalized anxiety (Hospital Anxiety and Depression Scale) and social anxiety (Liebowitz Social Anxiety Scale) were assessed. Dystonia severity (Burke-Fahn-Marsden Dystonia Rating Scale) and dystonic tremor were examined. Statistical predictors of HR-QoL were calculated using saturated path analysis.
Results: Reduced HR-QoL was strongly associated with the degree of depressive symptoms and generalized and social anxiety (8/8 RAND36 subscales, p ≤ .001). Increased dystonia severity was associated with worse physical functioning, physical and emotional role functioning, and social functioning (all p ≤ .001). The presence of tremor correlated with worse physical functioning and pain (all p ≤ .006). Younger age was associated with reduced emotional well-being and vitality (all p ≤ .006). There were no HR-QoL differences between sexes.
Conclusion: HR-QoL in isolated dystonia is strongly associated with psychiatric and physical features. While current standard of care focus on motor aspects of dystonia, comprehensive care should address both physical and mental aspects of health.

Text
JNNP_2020_325193_2020.12.23_highlighted - Accepted Manuscript
Download (473kB)

More information

Accepted/In Press date: 29 December 2020
e-pub ahead of print date: 9 February 2021

Identifiers

Local EPrints ID: 446275
URI: http://eprints.soton.ac.uk/id/eprint/446275
ISSN: 0022-3050
PURE UUID: 38d6221a-cfa2-4c6a-bee3-faec94631109
ORCID for J. Hall: ORCID iD orcid.org/0000-0001-8002-0922

Catalogue record

Date deposited: 03 Feb 2021 17:34
Last modified: 10 Jan 2022 03:11

Export record

Altmetrics

Contributors

Author: J. Junker
Author: B.D. Berman
Author: J. Hall ORCID iD
Author: D. Wahba
Author: V. Brandt
Author: J.S. Perlmutter
Author: J. Jankovic
Author: I.A. Malaty
Author: A.W. Shukla
Author: S.G. Reich
Author: A.J. Espay
Author: K.R. Duque
Author: N. Patel
Author: E. Roze
Author: M. Vidailhet
Author: H.A. Jinnah
Author: N. Bruegemann

Download statistics

Downloads from ePrints over the past year. Other digital versions may also be available to download e.g. from the publisher's website.

View more statistics

Atom RSS 1.0 RSS 2.0

Contact ePrints Soton: eprints@soton.ac.uk

ePrints Soton supports OAI 2.0 with a base URL of http://eprints.soton.ac.uk/cgi/oai2

This repository has been built using EPrints software, developed at the University of Southampton, but available to everyone to use.

We use cookies to ensure that we give you the best experience on our website. If you continue without changing your settings, we will assume that you are happy to receive cookies on the University of Southampton website.

×