An open-label trial in Friedreich ataxia suggests clinical benefit with high-dose resveratrol, without effect on frataxin levels

EM Yiu; G Tai; RE Peverill; KJ Lee; KD Croft; TA Mori; B Scheiber-Mojdehkar; B Sturm; M Praschberger; AP Vogel; G Rance; SEM Stephenson; JP Sarsero; C Stockley; CYJ Lee; A Churchyard; MV Evans-Galea; MM Ryan; PJ Lockhart; LA Corben; MB Delatycki

Journal article

An open-label trial in Friedreich ataxia suggests clinical benefit with high-dose resveratrol, without effect on frataxin levels

EM Yiu, G Tai, RE Peverill, KJ Lee, KD Croft, TA Mori, B Scheiber-Mojdehkar, B Sturm, M Praschberger, AP Vogel, G Rance, SEM Stephenson, JP Sarsero, C Stockley, CYJ Lee, A Churchyard, MV Evans-Galea, MM Ryan, PJ Lockhart, LA Corben Show all

Journal of Neurology | SPRINGER HEIDELBERG | Published : 2015

DOI: 10.1007/s00415-015-7719-2

Abstract

Friedreich ataxia (FRDA) is due to a triplet repeat expansion in FXN, resulting in deficiency of the mitochondrial protein frataxin. Resveratrol is a naturally occurring polyphenol, identified to increase frataxin expression in cellular and mouse models of FRDA and has anti-oxidant properties. This open-label, non-randomized trial evaluated the effect of two different doses of resveratrol on peripheral blood mononuclear cell (PBMC) frataxin levels over a 12-week period in individuals with FRDA. Secondary outcome measures included PMBC FXN mRNA, oxidative stress markers, and clinical measures of disease severity. Safety and tolerability were studied. Twenty-four participants completed the stu..

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Funding Acknowledgements

This study was funded by the Kyle Bryant Translational Research Award, Friedreich Ataxia Research Alliance (USA). The authors gratefully acknowledge the participants of this study. This work was made possible through Victorian State Government Operational Infrastructure Support and Australian Government NHMRC IRIISS.