A randomized, controlled, double-blind, crossover trial of zonisamide in myoclonus-dystonia - Université de Rennes Accéder directement au contenu
Article Dans Une Revue Neurology Année : 2016

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

Gérald Glibert
  • Fonction : Auteur
Anne Doe de Maindreville
  • Fonction : Auteur
Thibaud Lebouvier
  • Fonction : Auteur
David Grabli
André-Pierre Legrand
  • Fonction : Auteur
Anne Bissery
  • Fonction : Auteur
Alain Mallet
  • Fonction : Auteur

Résumé

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. Classification of evidence: This study provides Class I evidence that zonisamide improves myoclonus and related disability in patients with myoclonus-dystonia
Fichier non déposé

Dates et versions

hal-01302410 , version 1 (14-04-2016)

Identifiants

Citer

Elodie Hainque, Marie Vidailhet, Nathalie Cozic, Fanny Charbonnier-Beaupel, Stephane Thobois, et al.. A randomized, controlled, double-blind, crossover trial of zonisamide in myoclonus-dystonia. Neurology, 2016, 86 (18), pp.1729-35. ⟨10.1212/WNL.0000000000002631⟩. ⟨hal-01302410⟩
164 Consultations
0 Téléchargements

Altmetric

Partager

Gmail Facebook X LinkedIn More