c-Abl Regulates the Pathological Deposition of TDP-43 via Tyrosine 43 Phosphorylation

Cells. 2022 Dec 8;11(24):3972. doi: 10.3390/cells11243972.

Abstract

Non-receptor tyrosine kinase, c-Abl plays a role in the pathogenesis of several neurodegenerative disorders such as Alzheimer's disease and Parkinson's disease. Here, we found that TDP-43, which was one of the main proteins comprising pathological deposits in amyotrophic lateral sclerosis (ALS), is a novel substrate for c-Abl. The phosphorylation of tyrosine 43 of TDP-43 by c-Abl led to increased TDP-43 levels in the cytoplasm and increased the formation of G3BP1-positive stress granules in SH-SY5Y cells. The kinase-dead mutant of c-Abl had no effect on the cytoplasmic localization of TDP-43. The expression of phosphor-mimetic mutant Y43E of TDP-43 in primary cortical neurons accumulated the neurite granule. Furthermore, the phosphorylation of TDP-43 at tyrosine 43 by c-Abl promoted the aggregation of TDP-43 and increased neuronal cell death in primary cortical neurons, but not in c-Abl-deficient primary cortical neurons. Identification of c-Abl as the kinase of TDP43 provides new insight into the pathogenesis of ALS.

Keywords: TDP-43; amyotrophic lateral sclerosis; c-Abl; mislocalization; phosphorylation.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, N.I.H., Extramural

MeSH terms

  • Amyotrophic Lateral Sclerosis* / genetics
  • Amyotrophic Lateral Sclerosis* / metabolism
  • Amyotrophic Lateral Sclerosis* / pathology
  • DNA Helicases / metabolism
  • DNA-Binding Proteins / genetics
  • DNA-Binding Proteins / metabolism
  • Humans
  • Neuroblastoma
  • Phosphorylation
  • Poly-ADP-Ribose Binding Proteins / metabolism
  • Proto-Oncogene Proteins c-abl* / genetics
  • Proto-Oncogene Proteins c-abl* / metabolism
  • RNA Helicases / metabolism
  • RNA Recognition Motif Proteins / metabolism
  • Tyrosine / metabolism

Substances

  • DNA Helicases
  • DNA-Binding Proteins
  • G3BP1 protein, human
  • Poly-ADP-Ribose Binding Proteins
  • RNA Helicases
  • RNA Recognition Motif Proteins
  • Proto-Oncogene Proteins c-abl
  • TARDBP protein, human
  • Tyrosine