The mTOR Signaling Pathway in Multiple Sclerosis; from Animal Models to Human Data

Int J Mol Sci. 2022 Jul 22;23(15):8077. doi: 10.3390/ijms23158077.

Abstract

This article recapitulates the evidence on the role of mammalian targets of rapamycin (mTOR) complex pathways in multiple sclerosis (MS). Key biological processes that intersect with mTOR signaling cascades include autophagy, inflammasome activation, innate (e.g., microglial) and adaptive (B and T cell) immune responses, and axonal and neuronal toxicity/degeneration. There is robust evidence that mTOR inhibitors, such as rapamycin, ameliorate the clinical course of the animal model of MS, experimental autoimmune encephalomyelitis (EAE). New, evolving data unravel mechanisms underlying the therapeutic effect on EAE, which include balance among T-effector and T-regulatory cells, and mTOR effects on myeloid cell function, polarization, and antigen presentation, with relevance to MS pathogenesis. Radiologic and preliminary clinical data from a phase 2 randomized, controlled trial of temsirolimus (a rapamycin analogue) in MS show moderate efficacy, with significant adverse effects. Large clinical trials of indirect mTOR inhibitors (metformin) in MS are lacking; however, a smaller prospective, non-randomized study shows some potentially promising radiological results in combination with ex vivo beneficial effects on immune cells that might warrant further investigation. Importantly, the study of mTOR pathway contributions to autoimmune inflammatory demyelination and multiple sclerosis illustrates the difficulties in the clinical application of animal model results. Nevertheless, it is not inconceivable that targeting metabolism in the future with cell-selective mTOR inhibitors (compared to the broad inhibitors tried to date) could be developed to improve efficacy and reduce side effects.

Keywords: mTOR; metformin; multiple sclerosis; rapamycin.

Publication types

  • Review

MeSH terms

  • Animals
  • Clinical Trials, Phase II as Topic
  • Disease Models, Animal
  • Encephalomyelitis, Autoimmune, Experimental* / drug therapy
  • Humans
  • Mice
  • Mice, Inbred C57BL
  • Multiple Sclerosis* / pathology
  • Prospective Studies
  • Randomized Controlled Trials as Topic
  • Signal Transduction
  • Sirolimus / pharmacology
  • Sirolimus / therapeutic use
  • TOR Serine-Threonine Kinases* / metabolism

Substances

  • MTOR protein, human
  • TOR Serine-Threonine Kinases
  • Sirolimus

Grants and funding

LS is the site investigator in the trials MUSETTE (BN42082) and GAVOTTE (BN42083), sponsored by F. Hoffmann La-Roche Ltd. PS has received travel grants from Sanofi and support and research funding from the Onassis Foundation. AGV, MEB, and MA have nothing to disclose in relation to the present study.