articleNew England Journal of MedicineMay 7, 2019BRONZE OA

Targeting Huntingtin Expression in Patients with Huntington’s Disease

SJSarah J. TabriziBRBlair R. LeavittGBG. Bernhard LandwehrmeyerEJEdward J. WildCSCarsten Saft

UK Dementia Research Institute · University College London

PubMed
Indexed incrossrefpubmed

Abstract

Background

messenger RNA and thereby reduce concentrations of mutant huntingtin.

Methods

pharmacokinetics in cerebrospinal fluid (CSF). Prespecified exploratory end points included the concentration of mutant huntingtin in CSF.

Results

10-mg, 30-mg, 60-mg, 90-mg, and 120-mg dose groups, respectively).

Conclusions

to patients with early Huntington's disease was not accompanied by serious adverse events. We observed dose-dependent reductions in concentrations of mutant huntingtin. (Funded by Ionis Pharmaceuticals and F. Hoffmann-La Roche; ClinicalTrials.gov number, NCT02519036.).

Citation impact

669
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Authors

22
  • SJ
    Sarah J. TabriziCorresponding

    UK Dementia Research Institute, University College London

  • BR
    Blair R. Leavitt

    UK Dementia Research Institute, University College London

  • GB
    G. Bernhard Landwehrmeyer

    UK Dementia Research Institute, University College London

  • EJ
    Edward J. Wild

    UK Dementia Research Institute, University College London

  • CS
    Carsten Saft

    UK Dementia Research Institute, University College London

Topics & keywords

Keywords
  • Huntingtin
  • Disease
  • Adverse effect
  • Mutant
  • Mutation
  • Lysosomal storage disease
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