Titre
A randomized, controlled, double-blind, crossover trial of zonisamide in myoclonus-dystonia.
Type
article
Institution
Externe
Périodique
Auteur(s)
Hainque, E.
Auteure/Auteur
Vidailhet, M.
Auteure/Auteur
Cozic, N.
Auteure/Auteur
Charbonnier-Beaupel, F.
Auteure/Auteur
Thobois, S.
Auteure/Auteur
Tranchant, C.
Auteure/Auteur
Brochard, V.
Auteure/Auteur
Glibert, G.
Auteure/Auteur
Drapier, S.
Auteure/Auteur
Mutez, E.
Auteure/Auteur
Doe De Maindreville, A.
Auteure/Auteur
Lebouvier, T.
Auteure/Auteur
Hubsch, C.
Auteure/Auteur
Degos, B.
Auteure/Auteur
Bonnet, C.
Auteure/Auteur
Grabli, D.
Auteure/Auteur
Legrand, A.P.
Auteure/Auteur
Méneret, A.
Auteure/Auteur
Azulay, J.P.
Auteure/Auteur
Bissery, A.
Auteure/Auteur
Zahr, N.
Auteure/Auteur
Clot, F.
Auteure/Auteur
Mallet, A.
Auteure/Auteur
Dupont, S.
Auteure/Auteur
Apartis, E.
Auteure/Auteur
Corvol, J.C.
Auteure/Auteur
Roze, E.
Auteure/Auteur
Liens vers les personnes
ISSN
1526-632X
Statut éditorial
Publié
Date de publication
2016-05-03
Volume
86
Numéro
18
Première page
1729
Dernière page/numéro d’article
1735
Peer-reviewed
Oui
Langue
anglais
Notes
Publication types: Journal Article ; Randomized Controlled Trial
Publication Status: ppublish
Publication Status: ppublish
Résumé
To evaluate the efficacy and safety of zonisamide in patients with myoclonus-dystonia.
We conducted a randomized, double-blind, placebo-controlled crossover trial of zonisamide (300 mg/d) in 24 patients with myoclonus-dystonia. Each treatment period consisted of a 6-week titration phase followed by a 3-week fixed-dose phase. The periods were separated by a 5-week washout period. The co-primary outcomes were action myoclonus severity (section 4 of the Unified Myoclonus Rating Scale [UMRS 4]) and myoclonus-related functional disability (UMRS 5). Secondary outcomes included dystonia severity, assessed with the movement and disability subscales of the Burke-Fahn-Marsden-Dystonia Rating Scale (BFM), the Clinical Global Impression-Improvement scale (CGI), and safety measures. Wilcoxon signed-rank tests for paired data were used to analyze treatment effects.
Twenty-three patients (11 men, 12 women) were analyzed in the intention-to-treat analysis. Zonisamide significantly improved both action myoclonus (median improvement [95% confidence limits] -5 [-9.25 to -1.44], p = 0.003) and myoclonus-related functional disability (median improvement [95% confidence limits] -2 [-2.58 to -2.46], p = 0.007) compared to placebo. Zonisamide also significantly improved dystonia (BFM movement) compared to placebo (median improvement [95% confidence limits] -3 [-8.46 to 0.03], p = 0.009). No difference was found between zonisamide and placebo with respect to the CGI (median improvement [95% confidence limits] -1 [-1.31 to 0.09], p = 0.1). Zonisamide was well-tolerated.
Zonisamide is well-tolerated and effective on the motor symptoms of myoclonus-dystonia.
This study provides Class I evidence that zonisamide improves myoclonus and related disability in patients with myoclonus-dystonia.
We conducted a randomized, double-blind, placebo-controlled crossover trial of zonisamide (300 mg/d) in 24 patients with myoclonus-dystonia. Each treatment period consisted of a 6-week titration phase followed by a 3-week fixed-dose phase. The periods were separated by a 5-week washout period. The co-primary outcomes were action myoclonus severity (section 4 of the Unified Myoclonus Rating Scale [UMRS 4]) and myoclonus-related functional disability (UMRS 5). Secondary outcomes included dystonia severity, assessed with the movement and disability subscales of the Burke-Fahn-Marsden-Dystonia Rating Scale (BFM), the Clinical Global Impression-Improvement scale (CGI), and safety measures. Wilcoxon signed-rank tests for paired data were used to analyze treatment effects.
Twenty-three patients (11 men, 12 women) were analyzed in the intention-to-treat analysis. Zonisamide significantly improved both action myoclonus (median improvement [95% confidence limits] -5 [-9.25 to -1.44], p = 0.003) and myoclonus-related functional disability (median improvement [95% confidence limits] -2 [-2.58 to -2.46], p = 0.007) compared to placebo. Zonisamide also significantly improved dystonia (BFM movement) compared to placebo (median improvement [95% confidence limits] -3 [-8.46 to 0.03], p = 0.009). No difference was found between zonisamide and placebo with respect to the CGI (median improvement [95% confidence limits] -1 [-1.31 to 0.09], p = 0.1). Zonisamide was well-tolerated.
Zonisamide is well-tolerated and effective on the motor symptoms of myoclonus-dystonia.
This study provides Class I evidence that zonisamide improves myoclonus and related disability in patients with myoclonus-dystonia.
Sujets
PID Serval
serval:BIB_BE81214BE234
PMID
Date de création
2025-04-17T09:21:23.672Z
Date de création dans IRIS
2025-05-20T23:18:26Z