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March 11, 2014; 82 (10) Editorial

Feasibility of Huntington disease trials in the disease prodrome

Elizabeth A. McCusker, Richard H. Myers
First published February 7, 2014, DOI: https://doi.org/10.1212/WNL.0000000000000197
Elizabeth A. McCusker
From the Department of Neurology (E.A.M.), Westmead Hospital, and Sydney Medical School, University of Sydney, Australia; and Department of Neurology (R.H.M), Genome Science Institute, Boston University School of Medicine, Boston, MA.
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Richard H. Myers
From the Department of Neurology (E.A.M.), Westmead Hospital, and Sydney Medical School, University of Sydney, Australia; and Department of Neurology (R.H.M), Genome Science Institute, Boston University School of Medicine, Boston, MA.
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Feasibility of Huntington disease trials in the disease prodrome
Elizabeth A. McCusker, Richard H. Myers
Neurology Mar 2014, 82 (10) 824-825; DOI: 10.1212/WNL.0000000000000197

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In this issue of Neurology®, Rosas et al.1 present a phase II trial of creatine in Huntington disease (HD). This study is remarkable not only for the demonstration of safety and tolerability but also because the study design establishes the feasibility of clinical trials among premanifest HD individuals. Notably, the trial is undertaken in the disease prodrome, when the participants are clinically unaffected by HD, rather than in established disease. Furthermore, not all participants knew their mutation carrier status. The trial design defines methods to preserve confidentiality and overcomes the difficulty of studying HD at its earliest phase by avoiding the requirement to include only the genetically tested. The success of this study demonstrates that sufficient discriminant power can be obtained without the exclusion of genetically untested at-risk individuals.

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  • Go to Neurology.org for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the editorial.

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  • © 2014 American Academy of Neurology
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