Safety, pharmacokinetic, and functional effects of the nogo-a monoclonal antibody in amyotrophic lateral sclerosis: a randomized, first-in-human clinical trial

Vincent Meininger, Pierre-François Pradat, Andrea Corse, Safa Al-Sarraj, Benjamin Rix Brooks, James B Caress, Merit Cudkowicz, Stephen J Kolb, Dale Lange, P Nigel Leigh, Thomas Meyer, Stefano Milleri, Karen Morrison, Richard W Orrell, Gary Peters, Jeffrey D Rothstein, Jeremy Shefner, Arseniy Lavrov, Nicola Williams, Phil OverendJeffrey Price, Stewart Bates, Jonathan Bullman, David Krull, Alienor Berges, Bams Abila, Guy Meno-Tetang, Jens Wurthner

Research output: Contribution to journalArticlepeer-review

31 Citations (Scopus)

Abstract

UNLABELLED: The neurite outgrowth inhibitor, Nogo-A, has been shown to be overexpressed in skeletal muscle in amyotrophic lateral sclerosis (ALS); it is both a potential biomarker and therapeutic target. We performed a double-blind, two-part, dose-escalation study, in subjects with ALS, assessing safety, pharmacokinetics (PK) and functional effects of ozanezumab, a humanized monoclonal antibody against Nogo-A. In Part 1, 40 subjects were randomized (3∶1) to receive single dose intravenous ozanezumab (0.01, 0.1, 1, 5, or 15 mg/kg) or placebo. In Part 2, 36 subjects were randomized (3∶1) to receive two repeat doses of intravenous ozanezumab (0.5, 2.5, or 15 mg/kg) or placebo, approximately 4 weeks apart. The primary endpoints were safety and tolerability (adverse events [AEs], vital signs, electrocardiogram (ECG), and clinical laboratory tests). Secondary endpoints included PK, immunogenicity, functional endpoints (clinical and electrophysiological), and biomarker parameters. Overall, ozanezumab treatment (0.01-15 mg/kg) was well tolerated. The overall incidence of AEs in the repeat dose 2.5 mg/kg and 15 mg/kg ozanezumab groups was higher than in the repeat dose placebo group and repeat dose 0.5 mg/kg ozanezumab group. The majority were considered not related to study drug by the investigators. Six serious AEs were reported in three subjects receiving ozanezumab; none were considered related to study drug. No study drug-related patterns were identified for ECG, laboratory, or vital signs parameters. One subject (repeat dose 15 mg/kg ozanezumab) showed a weak, positive anti-ozanezumab-antibody result. PK results were generally consistent with monoclonal antibody treatments. No apparent treatment effects were observed for functional endpoints or muscle biomarkers. Immunohistochemical staining showed dose-dependent co-localization of ozanezumab with Nogo-A in skeletal muscle. In conclusion, single and repeat dose ozanezumab treatment was well tolerated and demonstrated co-localization at the site of action. These findings support future studies with ozanezumab in ALS.

TRIAL REGISTRATION: ClinicalTrials.gov NCT00875446 GSK-ClinicalStudyRegister.com GSK ID 111330.

Original languageEnglish
Article number0097803
JournalPLoS ONE
Volume9
Issue number5
DOIs
Publication statusPublished - 2014

Keywords

  • Administration, Intravenous
  • Amyotrophic Lateral Sclerosis
  • Antibodies, Monoclonal
  • Antibodies, Monoclonal, Humanized
  • Biological Markers
  • Dose-Response Relationship, Drug
  • Female
  • Humans
  • Immunohistochemistry
  • Male
  • Middle Aged
  • Myelin Proteins

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