Title :

A randomized, controlled, double-blind, crossover trial of zonisamide in myoclonus-dystonia

Author(s) :

Hainque, Elodie [Auteur]
Vidailhet, Marie [Auteur]
Cozic, Nathalie [Auteur]
Charbonnier-Beaupel, Fanny [Auteur]
Thobois, Stéphane [Auteur]
Tranchant, Christine [Auteur]
Brochard, Vanessa [Auteur]
Glibert, Gerald [Auteur]
Drapier, Sophie [Auteur]
Mutez, Eugenie [Auteur]


Doe De Maindreville, Anne [Auteur]
Lebouvier, Thibaud [Auteur]
Hubsch, Cecile [Auteur]
Degos, Bertrand [Auteur]
Bonnet, Cecilia [Auteur]
Grabli, David [Auteur]
Legrand, Andre-Pierre [Auteur]
Meneret, Aurelie [Auteur]
Azulay, Jean-Philippe [Auteur]
Bissery, Anne [Auteur]
Zahr, Noel [Auteur]
Clot, Fabienne [Auteur]
Mallet, Alain [Auteur]
Dupont, Sophie [Auteur]
Apartis, Emmanuelle [Auteur]
Corvol, Jean-Christophe [Auteur]
Roze, Emmanuel [Auteur]

Journal title :

Neurology

Abbreviated title :

Neurology

Volume number :

86

Pages :

1729-1735

Publication date :

2016-05-03

ISSN :

0028-3878

HAL domain(s) :

Sciences du Vivant [q-bio]

English abstract : [en]

OBJECTIVE: To evaluate the efficacy and safety of zonisamide in patients with myoclonus-dystonia. METHODS: We conducted a randomized, double-blind, placebo-controlled crossover trial of zonisamide (300 mg/d) in 24 patients ...
Show more >OBJECTIVE: To evaluate the efficacy and safety of zonisamide in patients with myoclonus-dystonia. METHODS: 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. RESULTS: 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. CONCLUSIONS: Zonisamide is well-tolerated and effective on the motor symptoms of myoclonus-dystonia. METHODS: This study provides Class I evidence that zonisamide improves myoclonus and related disability in patients with myoclonus-dystonia.Show less >

Language :

Anglais

Audience :

Internationale

Popular science :

Non

Administrative institution(s) :

CHU Lille
CNRS
Inserm
Université de Lille

Collections :

• Lille Neurosciences & Cognition (LilNCog) - U 1172

Submission date :

2019-11-27T14:31:09Z