A tuberous sclerosis complex signalling node at the peroxisome regulates mTORC1 and autophagy in response to ROS

Jiangwei Zhang, Jinhee Kim, Angela Alexander, Shengli Cai, Durga Nand Tripathi, Ruhee Dere, Andrew R. Tee, Jacqueline Tait-Mulder, Alessia Di Nardo, Juliette M. Han, Erica Kwiatkowski, Elaine A. Dunlop, Kayleigh M. Dodd, Rebecca D. Folkerth, Phyllis L. Faust, Michael B. Kastan, Mustafa Sahin, Cheryl Lyn Walker

Research output: Contribution to journalArticlepeer-review

213 Scopus citations

Abstract

Subcellular localization is emerging as an important mechanism for mTORC1 regulation. We report that the tuberous sclerosis complex (TSC) signalling node, TSC1, TSC2 and Rheb, localizes to peroxisomes, where it regulates mTORC1 in response to reactive oxygen species (ROS). TSC1 and TSC2 were bound by peroxisomal biogenesis factors 19 and 5 (PEX19 and PEX5), respectively, and peroxisome-localized TSC functioned as a Rheb GTPase-activating protein (GAP) to suppress mTORC1 and induce autophagy. Naturally occurring pathogenic mutations in TSC2 decreased PEX5 binding, and abrogated peroxisome localization, Rheb GAP activity and suppression of mTORC1 by ROS. Cells lacking peroxisomes were deficient in mTORC1 repression by ROS, and peroxisome-localization-deficient TSC2 mutants caused polarity defects and formation of multiple axons in neurons. These data identify a role for the TSC in responding to ROS at the peroxisome, and identify the peroxisome as a signalling organelle involved in regulation of mTORC1.

Original languageEnglish (US)
Pages (from-to)1186-1196
Number of pages11
JournalNature cell biology
Volume15
Issue number10
DOIs
StatePublished - Oct 2013

ASJC Scopus subject areas

  • Cell Biology

MD Anderson CCSG core facilities

  • High Resolution Electron Microscopy Facility

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