A myosin-7B-dependent endocytosis pathway mediates cellular entry of α-synuclein fibrils and polycation-bearing cargos

Proc Natl Acad Sci U S A. 2020 May 19;117(20):10865-10875. doi: 10.1073/pnas.1918617117. Epub 2020 May 4.

Abstract

Cell-to-cell transmission of misfolding-prone α-synuclein (α-Syn) has emerged as a key pathological event in Parkinson's disease. This process is initiated when α-Syn-bearing fibrils enter cells via clathrin-mediated endocytosis, but the underlying mechanisms are unclear. Using a CRISPR-mediated knockout screen, we identify SLC35B2 and myosin-7B (MYO7B) as critical endocytosis regulators for α-Syn preformed fibrils (PFFs). We show that SLC35B2, as a key regulator of heparan sulfate proteoglycan (HSPG) biosynthesis, is essential for recruiting α-Syn PFFs to the cell surface because this process is mediated by interactions between negatively charged sugar moieties of HSPGs and clustered K-T-K motifs in α-Syn PFFs. By contrast, MYO7B regulates α-Syn PFF cell entry by maintaining a plasma membrane-associated actin network that controls membrane dynamics. Without MYO7B or actin filaments, many clathrin-coated pits fail to be severed from the membrane, causing accumulation of large clathrin-containing "scars" on the cell surface. Intriguingly, the requirement for MYO7B in endocytosis is restricted to α-Syn PFFs and other polycation-bearing cargos that enter cells via HSPGs. Thus, our study not only defines regulatory factors for α-Syn PFF endocytosis, but also reveals a previously unknown endocytosis mechanism for HSPG-binding cargos in general, which requires forces generated by MYO7B and actin filaments.

Keywords: actin filament; clathrin-mediated endocytosis; heparan sulfate proteoglycan; myosin-7B; α-synuclein.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Cell Line
  • Clathrin / metabolism
  • Endocytosis / physiology*
  • Gene Knockout Techniques
  • HEK293 Cells
  • Humans
  • Models, Molecular
  • Myosins / chemistry*
  • Myosins / metabolism*
  • Parkinson Disease / metabolism
  • Polyelectrolytes / metabolism*
  • Protein Conformation
  • Sulfate Transporters / genetics
  • Sulfate Transporters / metabolism
  • alpha-Synuclein / metabolism*

Substances

  • Clathrin
  • Polyelectrolytes
  • SLC35B2 protein, human
  • Sulfate Transporters
  • alpha-Synuclein
  • polycations
  • MYO7B protein, human
  • Myosins