eprintid: 10071682
rev_number: 58
eprint_status: archive
userid: 608
dir: disk0/10/07/16/82
datestamp: 2019-04-08 09:51:29
lastmod: 2021-10-23 22:54:43
status_changed: 2019-04-08 09:51:29
type: article
metadata_visibility: show
creators_name: Mainka, T
creators_name: Erro, R
creators_name: Rothwell, J
creators_name: Kühn, AA
creators_name: Bhatia, KP
creators_name: Ganos, C
title: Remission in dystonia – Systematic review of the literature and meta-analysis
ispublished: pub
divisions: UCL
divisions: B02
divisions: C07
divisions: D07
divisions: F84
keywords: Blepharospasm, Dystonia, Recovery, Remission, Torticollis
note: This version is the author accepted manuscript. For information on re-use, please refer to the publisher’s terms and conditions.
abstract: In isolated, sporadic dystonia, it has been occasionally reported that some patients might undergo symptom remission. However, the exact clinical characteristics of patients with remission remain understudied. Given the important prognostic and pathophysiological implications of dystonic remission, we here provide a systematic review of the literature and a meta-analysis to assess demographic and clinical features associated with this phenomenon. We also provide a list of operational criteria to better define dystonic remission. Using PubMed and Embase, we conducted a systematic literature search in March 2018. 626 records were screened, 31 studies comprising data of 2551 cases with reports predominantly from patients with cervical dystonia (n = 1319) or blepharospasm/Meige syndrome (n = 704) were included in qualitative analysis. Five studies reporting remission in cervical dystonia were eligible for meta-analysis. Complete remission was reported in 11.8% and partial remission for 4.4% of cases. Remission rates were higher in cervical dystonia than in blepharospasm/Meige (e.g. complete remission 15.4% vs. 5.8% respectively). Remission occurred on average 4.5 years after onset of dystonic symptoms. However, the majority of patients (63.8%) relapsed. Meta-analysis for cervical dystonia showed that patients with remission were significantly younger at symptom onset than patients without remission (mean difference -7.13 years [95% CI: 10.58, -3.68], p < 0.0001). Based on our findings, we propose that the degree, the conditions associated with the onset, and the duration of remission are key factors to be considered in a unifying definition of dystonic remission.
date: 2019-09
date_type: published
official_url: https://doi.org/10.1016/j.parkreldis.2019.02.020
oa_status: green
full_text_type: other
language: eng
primo: open
primo_central: open_green
verified: verified_manual
elements_id: 1642376
doi: 10.1016/j.parkreldis.2019.02.020
pii: S1353-8020(19)30066-5
lyricists_name: Bhatia, Kailash
lyricists_name: Rothwell, John
lyricists_id: KPBHA96
lyricists_id: JCROT52
actors_name: Rothwell, John
actors_id: JCROT52
actors_role: owner
full_text_status: public
publication: Parkinsonism & Related Disorders
volume: 66
pagerange: 9-15
event_location: England
issn: 1873-5126
citation:        Mainka, T;    Erro, R;    Rothwell, J;    Kühn, AA;    Bhatia, KP;    Ganos, C;      (2019)    Remission in dystonia – Systematic review of the literature and meta-analysis.                   Parkinsonism & Related Disorders , 66    pp. 9-15.    10.1016/j.parkreldis.2019.02.020 <https://doi.org/10.1016/j.parkreldis.2019.02.020>.       Green open access   
 
document_url: https://discovery.ucl.ac.uk/id/eprint/10071682/24/Rothwell%20RemissionInDystonia_Final.pdf
document_url: https://discovery.ucl.ac.uk/id/eprint/10071682/9/Rothwell%20Figures_RemissionDystonia_20180813-tm.pdf
document_url: https://discovery.ucl.ac.uk/id/eprint/10071682/14/Rothwell%20Supplement1_20180813-tm.pdf
document_url: https://discovery.ucl.ac.uk/id/eprint/10071682/19/Rothwell%20Tables_RemissionDystonia_20180813-tm.pdf