Multiple Nf1 Schwann cell populations reprogram the plexiform neurofibroma tumor microenvironment

JCI Insight. 2022 Sep 22;7(18):e154513. doi: 10.1172/jci.insight.154513.

Abstract

To define alterations early in tumor formation, we studied nerve tumors in neurofibromatosis 1 (NF1), a tumor predisposition syndrome. Affected individuals develop neurofibromas, benign tumors driven by NF1 loss in Schwann cells (SCs). By comparing normal nerve cells to plexiform neurofibroma (PN) cells using single-cell and bulk RNA sequencing, we identified changes in 5 SC populations, including a de novo SC progenitor-like (SCP-like) population. Long after Nf1 loss, SC populations developed PN-specific expression of Dcn, Postn, and Cd74, with sustained expression of the injury response gene Postn and showed dramatic expansion of immune and stromal cell populations; in corresponding human PNs, the immune and stromal cells comprised 90% of cells. Comparisons between injury-related and tumor monocytes/macrophages support early monocyte recruitment and aberrant macrophage differentiation. Cross-species analysis verified each SC population and unique conserved patterns of predicted cell-cell communication in each SC population. This analysis identified PROS1-AXL, FGF-FGFR, and MIF-CD74 and its effector pathway NF-κB as deregulated in NF1 SC populations, including SCP-like cells predicted to influence other types of SCs, stromal cells, and/or immune cells in mouse and human. These findings highlight remarkable changes in multiple types of SCs and identify therapeutic targets for PN.

Keywords: Cell Biology; Expression profiling; Genetic diseases; NF-kappaB; Oncology.

Publication types

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

MeSH terms

  • Animals
  • Humans
  • Mice
  • NF-kappa B / metabolism
  • Neurofibroma, Plexiform* / genetics
  • Neurofibroma, Plexiform* / metabolism
  • Neurofibroma, Plexiform* / pathology
  • Neurofibromatosis 1* / genetics
  • Neurofibromatosis 1* / pathology
  • Schwann Cells / metabolism
  • Schwann Cells / pathology
  • Tumor Microenvironment

Substances

  • NF-kappa B