An mTORC1-mediated negative feedback loop constrains amino acid-induced FLCN-Rag activation in renal cells with TSC2 loss

Abstract

The mechanistic target of rapamycin complex 1 (mTORC1) integrates inputs from growth factors and nutrients, but how mTORC1 autoregulates its activity remains unclear. The MiT/TFE transcription factors are phosphorylated and inactivated by mTORC1 following lysosomal recruitment by RagC/D GTPases in response to amino acid stimulation. We find that starvation-induced lysosomal localization of the RagC/D GAP complex, FLCN:FNIP2, is markedly impaired in a mTORC1-sensitive manner in renal cells with TSC2 loss, resulting in unexpected TFEB hypophosphorylation and activation upon feeding. TFEB phosphorylation in TSC2-null renal cells is partially restored by destabilization of the lysosomal folliculin complex (LFC) induced by FLCN mutants and is fully rescued by forced lysosomal localization of the FLCN:FNIP2 dimer. Our data indicate that a negative feedback loop constrains amino acid-induced, FLCN:FNIP2-mediated RagC activity in renal cells with constitutive mTORC1 signaling, and the resulting MiT/TFE hyperactivation may drive oncogenesis with loss of the TSC2 tumor suppressor.

Document Details

Document Type
Pub Defense Publication
Publication Date
Nov 10, 2022
Source ID
10.1038/s41467-022-34617-7

Entities

People

  • Adrianna A. Mendes
  • Brandon Lam
  • Clarence Rachel Villanueva
  • Daniela C Salles
  • Ethan Schaffer
  • Hans B Liu
  • Juhyung Woo
  • Kaushal Asrani
  • Kewen Feng
  • Lia Oliveira
  • Pedram Argani
  • Sanjana Murali
  • Tamara L Lotan
  • Thiago Vidotto

Organizations

  • Congressionally Directed Medical Research Programs
  • National Cancer Institute
  • United States Department of Health and Human Services

Tags

Fields of Study

  • Biology

Readers

  • Aquatic Ecology
  • Molecular Biology and Genetics
  • Molecular and Cellular Biology