articleNew England Journal of MedicineJul 8, 2020DEBRONZE OA

Phase 1–2 Trial of Antisense Oligonucleotide Tofersen for SOD1 ALS

Washington University in St. Louis · University of Sheffield · +21 more institutions

PubMed
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Abstract

Background

mutations.

Methods

mutations. In each dose cohort (20, 40, 60, or 100 mg), participants were randomly assigned in a 3:1 ratio to receive five doses of tofersen or placebo, administered intrathecally for 12 weeks. The primary outcomes were safety and pharmacokinetics. The secondary outcome was the change from baseline in the cerebrospinal fluid (CSF) SOD1 concentration at day 85. Clinical function and vital capacity were measured.

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