Merlin cooperates with neurofibromin and Spred1 to suppress the Ras-Erk pathway

Hum Mol Genet. 2021 Feb 4;29(23):3793-3806. doi: 10.1093/hmg/ddaa263.

Abstract

The Ras-Erk pathway is frequently overactivated in human tumors. Neurofibromatosis types 1 and 2 (NF1, NF2) are characterized by multiple tumors of Schwann cell origin. The NF1 tumor suppressor neurofibromin is a principal Ras-GAP accelerating Ras inactivation, whereas the NF2 tumor suppressor merlin is a scaffold protein coordinating multiple signaling pathways. We have previously reported that merlin interacts with Ras and p120RasGAP. Here, we show that merlin can also interact with the neurofibromin/Spred1 complex via merlin-binding sites present on both proteins. Further, merlin can directly bind to the Ras-binding domain (RBD) and the kinase domain (KiD) of Raf1. As the third component of the neurofibromin/Spred1 complex, merlin cannot increase the Ras-GAP activity; rather, it blocks Ras binding to Raf1 by functioning as a 'selective Ras barrier'. Merlin-deficient Schwann cells require the Ras-Erk pathway activity for proliferation. Accordingly, suppression of the Ras-Erk pathway likely contributes to merlin's tumor suppressor activity. Taken together, our results, and studies by others, support targeting or co-targeting of this pathway as a therapy for NF2 inactivation-related tumors.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cells, Cultured
  • Extracellular Signal-Regulated MAP Kinases / genetics
  • Extracellular Signal-Regulated MAP Kinases / metabolism*
  • Neurofibromin 1 / genetics
  • Neurofibromin 1 / metabolism*
  • Neurofibromin 2 / genetics
  • Neurofibromin 2 / metabolism*
  • Rats
  • Repressor Proteins / genetics
  • Repressor Proteins / metabolism*
  • Schwann Cells / metabolism
  • Schwann Cells / pathology*
  • ras Proteins / genetics
  • ras Proteins / metabolism*

Substances

  • Neurofibromin 1
  • Neurofibromin 2
  • Nf2 protein, mouse
  • Repressor Proteins
  • Spred1 protein, rat
  • Extracellular Signal-Regulated MAP Kinases
  • ras Proteins