Neurotoxic Astrocytes Directly Converted from Sporadic and Familial ALS Patient Fibroblasts Reveal Signature Diversities and miR-146a Theragnostic Potential in Specific Subtypes

Cells. 2022 Apr 1;11(7):1186. doi: 10.3390/cells11071186.

Abstract

A lack of stratification methods in patients with amyotrophic lateral sclerosis (ALS) is likely implicated in therapeutic failures. Regional diversities and pathophysiological abnormalities in astrocytes from mice with SOD1 mutations (mSOD1-ALS) can now be explored in human patients using somatic cell reprogramming. Here, fibroblasts from four sporadic (sALS) and three mSOD1-ALS patients were transdifferentiated into induced astrocytes (iAstrocytes). ALS iAstrocytes were neurotoxic toward HB9-GFP mouse motor neurons (MNs) and exhibited subtype stratification through GFAP, CX43, Ki-67, miR-155 and miR-146a expression levels. Up- (two cases) and down-regulated (three cases) miR-146a values in iAstrocytes were recapitulated in their secretome, either free or as cargo in small extracellular vesicles (sEVs). We previously showed that the neuroprotective phenotype of depleted miR-146 mSOD1 cortical astrocytes was reverted by its mimic. Thus, we tested such modulation in the most miR-146a-depleted patient-iAstrocytes (one sALS and one mSOD1-ALS). The miR-146a mimic in ALS iAstrocytes counteracted their reactive/inflammatory profile and restored miR-146a levels in sEVs. A reduction in lysosomal activity and enhanced synaptic/axonal transport-related genes in NSC-34 MNs occurred after co-culture with miR-146a-modulated iAstrocytes. In summary, the regulation of miR-146a in depleted ALS astrocytes may be key in reestablishing their normal function and in restoring MN lysosomal/synaptic dynamic plasticity in disease sub-groups.

Keywords: amyotrophic lateral sclerosis (ALS); astrocyte-mediated neurotoxicity; fibroblast-transdifferentiated astrocytes; inflammatory-associated miRNAs; miR-146a modulation; mutant SOD1 (mSOD1); patient phenotype subtypes; small extracellular vesicles (sEVs); sporadic ALS (sALS).

MeSH terms

  • Amyotrophic Lateral Sclerosis* / genetics
  • Animals
  • Astrocytes
  • Disease Models, Animal
  • Fibroblasts
  • Humans
  • Mice
  • MicroRNAs* / genetics
  • Neurotoxicity Syndromes*

Substances

  • MIRN146 microRNA, human
  • MicroRNAs

Supplementary concepts

  • Amyotrophic lateral sclerosis 1