Spatial and functional separation of mTORC1 signalling in response to different amino acid sources.


Journal

Nature cell biology
ISSN: 1476-4679
Titre abrégé: Nat Cell Biol
Pays: England
ID NLM: 100890575

Informations de publication

Date de publication:
09 Oct 2024
Historique:
received: 01 02 2024
accepted: 09 09 2024
medline: 10 10 2024
pubmed: 10 10 2024
entrez: 9 10 2024
Statut: aheadofprint

Résumé

Amino acid (AA) availability is a robust determinant of cell growth through controlling mechanistic/mammalian target of rapamycin complex 1 (mTORC1) activity. According to the predominant model in the field, AA sufficiency drives the recruitment and activation of mTORC1 on the lysosomal surface by the heterodimeric Rag GTPases, from where it coordinates the majority of cellular processes. Importantly, however, the teleonomy of the proposed lysosomal regulation of mTORC1 and where mTORC1 acts on its effector proteins remain enigmatic. Here, by using multiple pharmacological and genetic means to perturb the lysosomal AA-sensing and protein recycling machineries, we describe the spatial separation of mTORC1 regulation and downstream functions in mammalian cells, with lysosomal and non-lysosomal mTORC1 phosphorylating distinct substrates in response to different AA sources. Moreover, we reveal that a fraction of mTOR localizes at lysosomes owing to basal lysosomal proteolysis that locally supplies new AAs, even in cells grown in the presence of extracellular nutrients, whereas cytoplasmic mTORC1 is regulated by exogenous AAs. Overall, our study substantially expands our knowledge about the topology of mTORC1 regulation by AAs and hints at the existence of distinct, Rag- and lysosome-independent mechanisms that control its activity at other subcellular locations. Given the importance of mTORC1 signalling and AA sensing for human ageing and disease, our findings will probably pave the way towards the identification of function-specific mTORC1 regulators and thus highlight more effective targets for drug discovery against conditions with dysregulated mTORC1 activity in the future.

Identifiants

pubmed: 39385049
doi: 10.1038/s41556-024-01523-7
pii: 10.1038/s41556-024-01523-7
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Subventions

Organisme : EC | EU Framework Programme for Research and Innovation H2020 | H2020 Priority Excellent Science | H2020 European Research Council (H2020 Excellent Science - European Research Council)
ID : 757729
Organisme : Deutsche Forschungsgemeinschaft (German Research Foundation)
ID : DE 3170/1-1
Organisme : Deutsche Forschungsgemeinschaft (German Research Foundation)
ID : DE 3170/1-1

Informations de copyright

© 2024. The Author(s).

Références

Laplante, M. & Sabatini, D. M. mTOR signaling in growth control and disease. Cell 149, 274–293 (2012).
pubmed: 22500797 pmcid: 3331679 doi: 10.1016/j.cell.2012.03.017
Proud, C. G. mTOR Signalling in health and disease. Biochem. Soc. Trans. 39, 431–436 (2011).
pubmed: 21428914 doi: 10.1042/BST0390431
Liko, D. & Hall, M. N. mTOR in health and in sickness. J. Mol. Med. 93, 1061–1073 (2015).
pubmed: 26391637 doi: 10.1007/s00109-015-1326-7
Valvezan, A. J. & Manning, B. D. Molecular logic of mTORC1 signalling as a metabolic rheostat. Nat. Metab. 1, 321–333 (2019).
pubmed: 32694720 doi: 10.1038/s42255-019-0038-7
Kennedy, B. K. & Lamming, D. W. The mechanistic target of rapamycin: the grand conductor of metabolism and aging. Cell Metab. 23, 990–1003 (2016).
pubmed: 27304501 pmcid: 4910876 doi: 10.1016/j.cmet.2016.05.009
Gonzalez, A. & Hall, M. N. Nutrient sensing and TOR signaling in yeast and mammals. EMBO J. 36, 397–408 (2017).
pubmed: 28096180 pmcid: 5694944 doi: 10.15252/embj.201696010
Liu, G. Y. & Sabatini, D. M. mTOR at the nexus of nutrition, growth, ageing and disease. Nat. Rev. Mol. Cell Biol. 21, 183–203 (2020).
pubmed: 31937935 pmcid: 7102936 doi: 10.1038/s41580-019-0199-y
Rabanal-Ruiz, Y. & Korolchuk, V. I. mTORC1 and nutrient homeostasis: the central role of the lysosome. Int. J. Mol. Sci. 19, 818 (2018).
pubmed: 29534520 pmcid: 5877679 doi: 10.3390/ijms19030818
Blommaart, E. F., Luiken, J. J., Blommaart, P. J., van Woerkom, G. M. & Meijer, A. J. Phosphorylation of ribosomal protein S6 is inhibitory for autophagy in isolated rat hepatocytes. J. Biol. Chem. 270, 2320–2326 (1995).
pubmed: 7836465 doi: 10.1074/jbc.270.5.2320
Hara, K. et al. Amino acid sufficiency and mTOR regulate p70 S6 kinase and eIF-4E BP1 through a common effector mechanism. J. Biol. Chem. 273, 14484–14494 (1998).
pubmed: 9603962 doi: 10.1074/jbc.273.23.14484
Efeyan, A., Zoncu, R. & Sabatini, D. M. Amino acids and mTORC1: from lysosomes to disease. Trends Mol. Med. 18, 524–533 (2012).
pubmed: 22749019 pmcid: 3432651 doi: 10.1016/j.molmed.2012.05.007
Jewell, J. L., Russell, R. C. & Guan, K. L. Amino acid signalling upstream of mTOR. Nat. Rev. Mol. Cell Biol. 14, 133–139 (2013).
pubmed: 23361334 pmcid: 3988467 doi: 10.1038/nrm3522
Fernandes, S. A. & Demetriades, C. The multifaceted role of nutrient sensing and mTORC1 signaling in physiology and aging. Front. Aging 2, 707372 (2021).
pubmed: 35822019 pmcid: 9261424 doi: 10.3389/fragi.2021.707372
Johnson, S. C., Rabinovitch, P. S. & Kaeberlein, M. mTOR is a key modulator of ageing and age-related disease. Nature 493, 338–345 (2013).
pubmed: 23325216 pmcid: 3687363 doi: 10.1038/nature11861
Cornu, M., Albert, V. & Hall, M. N. mTOR in aging, metabolism, and cancer. Curr. Opin. Genet. Dev. 23, 53–62 (2013).
pubmed: 23317514 doi: 10.1016/j.gde.2012.12.005
Tsang, C. K., Qi, H., Liu, L. F. & Zheng, X. F. Targeting mammalian target of rapamycin (mTOR) for health and diseases. Drug Discov. Today 12, 112–124 (2007).
pubmed: 17275731 doi: 10.1016/j.drudis.2006.12.008
Um, S. H., D’Alessio, D. & Thomas, G. Nutrient overload, insulin resistance, and ribosomal protein S6 kinase 1, S6K1. Cell Metab. 3, 393–402 (2006).
pubmed: 16753575 doi: 10.1016/j.cmet.2006.05.003
Sancak, Y. et al. The Rag GTPases bind raptor and mediate amino acid signaling to mTORC1. Science 320, 1496–1501 (2008).
pubmed: 18497260 pmcid: 2475333 doi: 10.1126/science.1157535
Kim, E., Goraksha-Hicks, P., Li, L., Neufeld, T. P. & Guan, K. L. Regulation of TORC1 by Rag GTPases in nutrient response. Nat. Cell Biol. 10, 935–945 (2008).
pubmed: 18604198 pmcid: 2711503 doi: 10.1038/ncb1753
Zoncu, R. et al. mTORC1 senses lysosomal amino acids through an inside-out mechanism that requires the vacuolar H(
pubmed: 22053050 pmcid: 3211112 doi: 10.1126/science.1207056
Gollwitzer, P., Grutzmacher, N., Wilhelm, S., Kummel, D. & Demetriades, C. A Rag GTPase dimer code defines the regulation of mTORC1 by amino acids. Nat. Cell Biol. 24, 1394–1406 (2022).
pubmed: 36097072 pmcid: 9481461 doi: 10.1038/s41556-022-00976-y
Sancak, Y. et al. Ragulator–Rag complex targets mTORC1 to the lysosomal surface and is necessary for its activation by amino acids. Cell 141, 290–303 (2010).
pubmed: 20381137 pmcid: 3024592 doi: 10.1016/j.cell.2010.02.024
Demetriades, C., Doumpas, N. & Teleman, A. A. Regulation of TORC1 in response to amino acid starvation via lysosomal recruitment of TSC2. Cell 156, 786–799 (2014).
pubmed: 24529380 pmcid: 4346203 doi: 10.1016/j.cell.2014.01.024
Powis, K. & De Virgilio, C. Conserved regulators of Rag GTPases orchestrate amino acid-dependent TORC1 signaling. Cell Discov. 2, 15049 (2016).
pubmed: 27462445 pmcid: 4860963 doi: 10.1038/celldisc.2015.49
Lim, C. Y. & Zoncu, R. The lysosome as a command-and-control center for cellular metabolism. J. Cell Biol. 214, 653–664 (2016).
pubmed: 27621362 pmcid: 5021098 doi: 10.1083/jcb.201607005
Betz, C. & Hall, M. N. Where is mTOR and what is it doing there? J. Cell Biol. 203, 563–574 (2013).
pubmed: 24385483 pmcid: 3840941 doi: 10.1083/jcb.201306041
Manifava, M. et al. Dynamics of mTORC1 activation in response to amino acids. eLife 5, e19960 (2016).
pubmed: 27725083 pmcid: 5059141 doi: 10.7554/eLife.19960
Lawrence, R. E. et al. A nutrient-induced affinity switch controls mTORC1 activation by its Rag GTPase–Ragulator lysosomal scaffold. Nat. Cell Biol. 20, 1052–1063 (2018).
pubmed: 30061680 pmcid: 6279252 doi: 10.1038/s41556-018-0148-6
Mauvezin, C. & Neufeld, T. P. Bafilomycin A1 disrupts autophagic flux by inhibiting both V-ATPase-dependent acidification and Ca-P60A/SERCA-dependent autophagosome-lysosome fusion. Autophagy 11, 1437–1438 (2015).
pubmed: 26156798 pmcid: 4590655 doi: 10.1080/15548627.2015.1066957
Musiwaro, P., Smith, M., Manifava, M., Walker, S. A. & Ktistakis, N. T. Characteristics and requirements of basal autophagy in HEK 293 cells. Autophagy 9, 1407–1417 (2013).
pubmed: 23800949 doi: 10.4161/auto.25455
Abu-Remaileh, M. et al. Lysosomal metabolomics reveals V-ATPase- and mTOR-dependent regulation of amino acid efflux from lysosomes. Science 358, 807–813 (2017).
pubmed: 29074583 pmcid: 5704967 doi: 10.1126/science.aan6298
Efeyan, A. et al. RagA, but not RagB, is essential for embryonic development and adult mice. Dev. Cell 29, 321–329 (2014).
pubmed: 24768164 pmcid: 4035553 doi: 10.1016/j.devcel.2014.03.017
Laqtom, N. N. et al. CLN3 is required for the clearance of glycerophosphodiesters from lysosomes. Nature 609, 1005–1011 (2022).
pubmed: 36131016 pmcid: 10510443 doi: 10.1038/s41586-022-05221-y
Rosner, M., Schipany, K. & Hengstschlager, M. p70 S6K1 nuclear localization depends on its mTOR-mediated phosphorylation at T389, but not on its kinase activity towards S6. Amino Acids 42, 2251–2256 (2012).
pubmed: 21710263 doi: 10.1007/s00726-011-0965-4
Pavan, I. C. et al. Different interactomes for p70-S6K1 and p54-S6K2 revealed by proteomic analysis. Proteomics 16, 2650–2666 (2016).
pubmed: 27493124 doi: 10.1002/pmic.201500249
Nuchel, J. et al. An mTORC1-GRASP55 signaling axis controls unconventional secretion to reshape the extracellular proteome upon stress. Mol. Cell. 81, 3275–3293 (2021).
pubmed: 34245671 pmcid: 8382303 doi: 10.1016/j.molcel.2021.06.017
Yang, G. et al. RagC phosphorylation autoregulates mTOR complex 1. EMBO J. 38, e99548 (2019).
pubmed: 30552228 doi: 10.15252/embj.201899548
Bar-Peled, L., Schweitzer, L. D., Zoncu, R. & Sabatini, D. M. Ragulator is a GEF for the rag GTPases that signal amino acid levels to mTORC1. Cell 150, 1196–1208 (2012).
pubmed: 22980980 pmcid: 3517996 doi: 10.1016/j.cell.2012.07.032
Demetriades, C., Plescher, M. & Teleman, A. A. Lysosomal recruitment of TSC2 is a universal response to cellular stress. Nat. Commun. 7, 10662 (2016).
pubmed: 26868506 pmcid: 4754342 doi: 10.1038/ncomms10662
Plescher, M., Teleman, A. A. & Demetriades, C. TSC2 mediates hyperosmotic stress-induced inactivation of mTORC1. Sci. Rep. 5, 13828 (2015).
pubmed: 26345496 pmcid: 4642562 doi: 10.1038/srep13828
Carroll, B. et al. Control of TSC2-Rheb signaling axis by arginine regulates mTORC1 activity. eLife 5, e11058 (2016).
pubmed: 26742086 pmcid: 4764560 doi: 10.7554/eLife.11058
Chantranupong, L. et al. The Sestrins interact with GATOR2 to negatively regulate the amino-acid-sensing pathway upstream of mTORC1. Cell Rep. 9, 1–8 (2014).
pubmed: 25263562 pmcid: 4223866 doi: 10.1016/j.celrep.2014.09.014
Chantranupong, L. et al. The CASTOR proteins are arginine sensors for the mTORC1 pathway. Cell 165, 153–164 (2016).
pubmed: 26972053 pmcid: 4808398 doi: 10.1016/j.cell.2016.02.035
Saxton, R. A. et al. Structural basis for leucine sensing by the Sestrin2-mTORC1 pathway. Science 351, 53–58 (2016).
pubmed: 26586190 doi: 10.1126/science.aad2087
Saxton, R. A., Chantranupong, L., Knockenhauer, K. E., Schwartz, T. U. & Sabatini, D. M. Mechanism of arginine sensing by CASTOR1 upstream of mTORC1. Nature 536, 229–233 (2016).
pubmed: 27487210 pmcid: 4988899 doi: 10.1038/nature19079
Parmigiani, A. et al. Sestrins inhibit mTORC1 kinase activation through the GATOR complex. Cell Rep. 9, 1281–1291 (2014).
pubmed: 25457612 pmcid: 4303546 doi: 10.1016/j.celrep.2014.10.019
Wolfson, R. L. et al. Sestrin2 is a leucine sensor for the mTORC1 pathway. Science 351, 43–48 (2016).
pubmed: 26449471 doi: 10.1126/science.aab2674
Chen, J. et al. SAR1B senses leucine levels to regulate mTORC1 signalling. Nature 596, 281–284 (2021).
pubmed: 34290409 doi: 10.1038/s41586-021-03768-w
Bar-Peled, L. et al. A tumor suppressor complex with GAP activity for the Rag GTPases that signal amino acid sufficiency to mTORC1. Science 340, 1100–1106 (2013).
pubmed: 23723238 pmcid: 3728654 doi: 10.1126/science.1232044
Panchaud, N., Peli-Gulli, M. P. & De Virgilio, C. Amino acid deprivation inhibits TORC1 through a GTPase-activating protein complex for the Rag family GTPase Gtr1. Sci. Signal 6, ra42 (2013).
pubmed: 23716719 doi: 10.1126/scisignal.2004112
Jewell, J. L. et al. Differential regulation of mTORC1 by leucine and glutamine. Science 347, 194–198 (2015).
pubmed: 25567907 pmcid: 4384888 doi: 10.1126/science.1259472
Meng, D. et al. Glutamine and asparagine activate mTORC1 independently of Rag GTPases. J. Biol. Chem. 295, 2890–2899 (2020).
pubmed: 32019866 pmcid: 7062167 doi: 10.1074/jbc.AC119.011578
Stracka, D., Jozefczuk, S., Rudroff, F., Sauer, U. & Hall, M. N. Nitrogen source activates TOR (target of rapamycin) complex 1 via glutamine and independently of Gtr/Rag proteins. J. Biol. Chem. 289, 25010–25020 (2014).
pubmed: 25063813 pmcid: 4155668 doi: 10.1074/jbc.M114.574335
Sardiello, M. et al. A gene network regulating lysosomal biogenesis and function. Science 325, 473–477 (2009).
pubmed: 19556463 doi: 10.1126/science.1174447
Settembre, C. et al. TFEB links autophagy to lysosomal biogenesis. Science 332, 1429–1433 (2011).
pubmed: 21617040 pmcid: 3638014 doi: 10.1126/science.1204592
Martina, J. A. et al. The nutrient-responsive transcription factor TFE3 promotes autophagy, lysosomal biogenesis, and clearance of cellular debris. Sci. Signal 7, ra9 (2014).
pubmed: 24448649 pmcid: 4696865 doi: 10.1126/scisignal.2004754
Puertollano, R., Ferguson, S. M., Brugarolas, J. & Ballabio, A. The complex relationship between TFEB transcription factor phosphorylation and subcellular localization. EMBO J. 37, e98804 (2018).
pubmed: 29764979 pmcid: 5983138 doi: 10.15252/embj.201798804
Duvel, K. et al. Activation of a metabolic gene regulatory network downstream of mTOR complex 1. Mol. Cell 39, 171–183 (2010).
pubmed: 20670887 pmcid: 2946786 doi: 10.1016/j.molcel.2010.06.022
Ben-Sahra, I., Hoxhaj, G., Ricoult, S. J. H., Asara, J. M. & Manning, B. D. mTORC1 induces purine synthesis through control of the mitochondrial tetrahydrofolate cycle. Science 351, 728–733 (2016).
pubmed: 26912861 pmcid: 4786372 doi: 10.1126/science.aad0489
Artoni, F., Grutzmacher, N. & Demetriades, C. Unbiased evaluation of rapamycin’s specificity as an mTOR inhibitor. Aging Cell 22, e13888 (2023).
pubmed: 37222020 pmcid: 10410055 doi: 10.1111/acel.13888
Settembre, C. et al. A lysosome-to-nucleus signalling mechanism senses and regulates the lysosome via mTOR and TFEB. EMBO J. 31, 1095–1108 (2012).
pubmed: 22343943 pmcid: 3298007 doi: 10.1038/emboj.2012.32
Thedieck, K. et al. Inhibition of mTORC1 by astrin and stress granules prevents apoptosis in cancer cells. Cell 154, 859–874 (2013).
pubmed: 23953116 doi: 10.1016/j.cell.2013.07.031
Averous, J. et al. Requirement for lysosomal localization of mTOR for its activation differs between leucine and other amino acids. Cell Signal 26, 1918–1927 (2014).
pubmed: 24793303 doi: 10.1016/j.cellsig.2014.04.019
Wang, A. et al. Activity-independent targeting of mTOR to lysosomes in primary osteoclasts. Sci. Rep. 7, 3005 (2017).
pubmed: 28592812 pmcid: 5462732 doi: 10.1038/s41598-017-03494-2
Oshiro, N., Rapley, J. & Avruch, J. Amino acids activate mammalian target of rapamycin (mTOR) complex 1 without changing Rag GTPase guanyl nucleotide charging. J. Biol. Chem. 289, 2658–2674 (2014).
pubmed: 24337580 doi: 10.1074/jbc.M113.528505
Buerger, C., DeVries, B. & Stambolic, V. Localization of Rheb to the endomembrane is critical for its signaling function. Biochem. Biophys. Res. Commun. 344, 869–880 (2006).
pubmed: 16631613 doi: 10.1016/j.bbrc.2006.03.220
Hanker, A. B. et al. Differential requirement of CAAX-mediated posttranslational processing for Rheb localization and signaling. Oncogene 29, 380–391 (2010).
pubmed: 19838215 doi: 10.1038/onc.2009.336
Yadav, R. B. et al. mTOR direct interactions with Rheb-GTPase and raptor: subcellular localization using fluorescence lifetime imaging. BMC Cell Biol. 14, 3 (2013).
pubmed: 23311891 pmcid: 3549280 doi: 10.1186/1471-2121-14-3
Hao, F. et al. Rheb localized on the Golgi membrane activates lysosome-localized mTORC1 at the Golgi–lysosome contact site. J. Cell Sci. 131, jcs208017 (2018).
pubmed: 29222112 doi: 10.1242/jcs.208017
Gosavi, P., Houghton, F. J., McMillan, P. J., Hanssen, E. & Gleeson, P. A. The Golgi ribbon in mammalian cells negatively regulates autophagy by modulating mTOR activity. J. Cell Sci. 131, jcs211987 (2018).
pubmed: 29361552 doi: 10.1242/jcs.211987
Angarola, B. & Ferguson, S. M. Weak membrane interactions allow Rheb to activate mTORC1 signaling without major lysosome enrichment. Mol. Biol. Cell 30, 2750–2760 (2019).
pubmed: 31532697 pmcid: 6789162 doi: 10.1091/mbc.E19-03-0146
Holz, M. K., Ballif, B. A., Gygi, S. P. & Blenis, J. mTOR and S6K1 mediate assembly of the translation preinitiation complex through dynamic protein interchange and ordered phosphorylation events. Cell 123, 569–580 (2005).
pubmed: 16286006 doi: 10.1016/j.cell.2005.10.024
Zhou, X. et al. Dynamic visualization of mTORC1 activity in living cells. Cell Rep. 10, 1767–1777 (2015).
pubmed: 25772363 pmcid: 4567530 doi: 10.1016/j.celrep.2015.02.031
Ahmed, A. R. et al. Direct imaging of the recruitment and phosphorylation of S6K1 in the mTORC1 pathway in living cells. Sci. Rep. 9, 3408 (2019).
pubmed: 30833605 pmcid: 6399282 doi: 10.1038/s41598-019-39410-z
Drenan, R. M., Liu, X., Bertram, P. G. & Zheng, X. F. FKBP12-rapamycin-associated protein or mammalian target of rapamycin (FRAP/mTOR) localization in the endoplasmic reticulum and the Golgi apparatus. J. Biol. Chem. 279, 772–778 (2004).
pubmed: 14578359 doi: 10.1074/jbc.M305912200
Liu, X. & Zheng, X. F. Endoplasmic reticulum and Golgi localization sequences for mammalian target of rapamycin. Mol. Biol. Cell 18, 1073–1082 (2007).
pubmed: 17215520 pmcid: 1805082 doi: 10.1091/mbc.e06-05-0406
Tsokanos, F. F. et al. eIF4A inactivates TORC1 in response to amino acid starvation. EMBO J. 35, 1058–1076 (2016).
pubmed: 26988032 pmcid: 4868951 doi: 10.15252/embj.201593118
Schieke, S. M. et al. The mammalian target of rapamycin (mTOR) pathway regulates mitochondrial oxygen consumption and oxidative capacity. J. Biol. Chem. 281, 27643–27652 (2006).
pubmed: 16847060 doi: 10.1074/jbc.M603536200
Mutvei, A. P. et al. Rap1-GTPases control mTORC1 activity by coordinating lysosome organization with amino acid availability. Nat. Commun. 11, 1416 (2020).
pubmed: 32184389 pmcid: 7078236 doi: 10.1038/s41467-020-15156-5
Lindquist, R. A. et al. Genome-scale RNAi on living-cell microarrays identifies novel regulators of Drosophila melanogaster TORC1-S6K pathway signaling. Genome Res. 21, 433–446 (2011).
pubmed: 21239477 pmcid: 3044857 doi: 10.1101/gr.111492.110
Teis, D. et al. p14-MP1-MEK1 signaling regulates endosomal traffic and cellular proliferation during tissue homeostasis. J. Cell Biol. 175, 861–868 (2006).
pubmed: 17178906 pmcid: 2064696 doi: 10.1083/jcb.200607025
Gangloff, Y. G. et al. Disruption of the mouse mTOR gene leads to early postimplantation lethality and prohibits embryonic stem cell development. Mol. Cell. Biol. 24, 9508–9516 (2004).
pubmed: 15485918 pmcid: 522282 doi: 10.1128/MCB.24.21.9508-9516.2004
Murakami, M. et al. mTOR is essential for growth and proliferation in early mouse embryos and embryonic stem cells. Mol. Cell. Biol. 24, 6710–6718 (2004).
pubmed: 15254238 pmcid: 444840 doi: 10.1128/MCB.24.15.6710-6718.2004
Guertin, D. A. et al. Ablation in mice of the mTORC components raptor, rictor, or mLST8 reveals that mTORC2 is required for signaling to Akt-FOXO and PKCalpha, but not S6K1. Dev. Cell 11, 859–871 (2006).
pubmed: 17141160 doi: 10.1016/j.devcel.2006.10.007
Kim, Y. C. et al. Rag GTPases are cardioprotective by regulating lysosomal function. Nat. Commun. 5, 4241 (2014).
pubmed: 24980141 doi: 10.1038/ncomms5241
Shen, K., Sidik, H. & Talbot, W. S. The Rag–Ragulator complex regulates lysosome function and phagocytic flux in microglia. Cell Rep. 14, 547–559 (2016).
pubmed: 26774477 pmcid: 4731305 doi: 10.1016/j.celrep.2015.12.055
Settembre, C. et al. TFEB controls cellular lipid metabolism through a starvation-induced autoregulatory loop. Nat. Cell Biol. 15, 647–658 (2013).
pubmed: 23604321 pmcid: 3699877 doi: 10.1038/ncb2718
Pastore, N. et al. TFE3 regulates whole-body energy metabolism in cooperation with TFEB. EMBO Mol. Med. 9, 605–621 (2017).
pubmed: 28283651 pmcid: 5412821 doi: 10.15252/emmm.201607204
Lawrence, R. E. et al. Structural mechanism of a Rag GTPase activation checkpoint by the lysosomal folliculin complex. Science 366, 971–977 (2019).
pubmed: 31672913 pmcid: 6945816 doi: 10.1126/science.aax0364
Napolitano, G. et al. A substrate-specific mTORC1 pathway underlies Birt–Hogg–Dube syndrome. Nature 585, 597–602 (2020).
pubmed: 32612235 pmcid: 7610377 doi: 10.1038/s41586-020-2444-0
Alesi, N. et al. TSC2 regulates lysosome biogenesis via a non-canonical RAGC and TFEB-dependent mechanism. Nat. Commun. 12, 4245 (2021).
pubmed: 34253722 pmcid: 8275687 doi: 10.1038/s41467-021-24499-6
Hatakeyama, R. et al. Spatially distinct pools of TORC1 balance protein homeostasis. Mol. Cell 73, 325–338 e328 (2019).
pubmed: 30527664 doi: 10.1016/j.molcel.2018.10.040
Mane, S. M. et al. Purification and characterization of human lysosomal membrane glycoproteins. Arch. Biochem. Biophys. 268, 360–378 (1989).
pubmed: 2912382 doi: 10.1016/0003-9861(89)90597-3
Chen, J. W., Murphy, T. L., Willingham, M. C., Pastan, I. & August, J. T. Identification of two lysosomal membrane glycoproteins. J. Cell Biol. 101, 85–95 (1985).
pubmed: 2409098 doi: 10.1083/jcb.101.1.85
Nuchel, J. et al. TGFB1 is secreted through an unconventional pathway dependent on the autophagic machinery and cytoskeletal regulators. Autophagy 14, 465–486 (2018).
pubmed: 29297744 pmcid: 5915026 doi: 10.1080/15548627.2017.1422850
Kowarz, E., Loscher, D. & Marschalek, R. Optimized Sleeping Beauty transposons rapidly generate stable transgenic cell lines. Biotechnol. J. 10, 647–653 (2015).
pubmed: 25650551 doi: 10.1002/biot.201400821
Ran, F. A. et al. Genome engineering using the CRISPR–Cas9 system. Nat. Protoc. 8, 2281–2308 (2013).
pubmed: 24157548 pmcid: 3969860 doi: 10.1038/nprot.2013.143
Wyant, G. A. et al. mTORC1 activator SLC38A9 is required to efflux essential amino acids from lysosomes and use protein as a nutrient. Cell 171, 642–654 e612 (2017).
pubmed: 29053970 pmcid: 5704964 doi: 10.1016/j.cell.2017.09.046
Mahoney, S. J. et al. A small molecule inhibitor of Rheb selectively targets mTORC1 signaling. Nat. Commun. 9, 548 (2018).
pubmed: 29416044 pmcid: 5803267 doi: 10.1038/s41467-018-03035-z
Sancak, Y. et al. PRAS40 is an insulin-regulated inhibitor of the mTORC1 protein kinase. Mol. Cell 25, 903–915 (2007).
pubmed: 17386266 doi: 10.1016/j.molcel.2007.03.003
Nicastro, R. et al. Malonyl-CoA is a conserved endogenous ATP-competitive mTORC1 inhibitor. Nat. Cell Biol. 25, 1303–1318 (2023).
pubmed: 37563253 pmcid: 10495264 doi: 10.1038/s41556-023-01198-6
Fitzian, K. et al. TSC1 binding to lysosomal PIPs is required for TSC complex translocation and mTORC1 regulation. Mol. Cell 81, 2705–2721 e2708 (2021).
pubmed: 33974911 doi: 10.1016/j.molcel.2021.04.019
Schindelin, J. et al. Fiji: an open-source platform for biological-image analysis. Nat. Methods 9, 676–682 (2012).
pubmed: 22743772 doi: 10.1038/nmeth.2019
Manders, E. M. M., Verbeek, F. J. & Aten, J. A. Measurement of co-localization of objects in dual-colour confocal images. J. Microsc. 169, 375–382 (1993).
pubmed: 33930978 doi: 10.1111/j.1365-2818.1993.tb03313.x
Costes, S. V. et al. Automatic and quantitative measurement of protein-protein colocalization in live cells. Biophys. J. 86, 3993–4003 (2004).
pubmed: 15189895 pmcid: 1304300 doi: 10.1529/biophysj.103.038422
Dunn, K. W., Kamocka, M. M. & McDonald, J. H. A practical guide to evaluating colocalization in biological microscopy. Am. J. Physiol. Cell Physiol. 300, C723–C742 (2011).
pubmed: 21209361 pmcid: 3074624 doi: 10.1152/ajpcell.00462.2010
Stirling, J. W. & Graff, P. S. Antigen unmasking for immunoelectron microscopy: labeling is improved by treating with sodium ethoxide or sodium metaperiodate, then heating on retrieval medium. J. Histochem. Cytochem. 43, 115–123 (1995).
pubmed: 7529784 doi: 10.1177/43.2.7529784
Rouillard, A. D. et al. The harmonizome: a collection of processed datasets gathered to serve and mine knowledge about genes and proteins. Database 2016, baw100 (2016).
pubmed: 27374120 pmcid: 4930834 doi: 10.1093/database/baw100
Igarashi, T. et al. Clock and ATF4 transcription system regulates drug resistance in human cancer cell lines. Oncogene 26, 4749–4760 (2007).
pubmed: 17297441 doi: 10.1038/sj.onc.1210289
Torrence, M. E. et al. The mTORC1-mediated activation of ATF4 promotes protein and glutathione synthesis downstream of growth signals. eLife 10, e63326 (2021).
pubmed: 33646118 pmcid: 7997658 doi: 10.7554/eLife.63326
Iqbal, A. et al. Flaski toolbox. GitHub https://flaski.age.mpg.de (2021).

Auteurs

Stephanie A Fernandes (SA)

Max Planck Institute for Biology of Ageing, Cologne, Germany.
Cologne Graduate School of Ageing Research, Cologne, Germany.

Danai-Dimitra Angelidaki (DD)

Max Planck Institute for Biology of Ageing, Cologne, Germany.

Julian Nüchel (J)

Max Planck Institute for Biology of Ageing, Cologne, Germany.
Center for Biochemistry, Medical Faculty, University of Cologne, Cologne, Germany.

Jiyoung Pan (J)

Max Planck Institute for Biology of Ageing, Cologne, Germany.
Cologne Graduate School of Ageing Research, Cologne, Germany.

Peter Gollwitzer (P)

Max Planck Institute for Biology of Ageing, Cologne, Germany.

Yoav Elkis (Y)

Max Planck Institute for Biology of Ageing, Cologne, Germany.

Filippo Artoni (F)

Max Planck Institute for Biology of Ageing, Cologne, Germany.
Cologne Graduate School of Ageing Research, Cologne, Germany.

Sabine Wilhelm (S)

Max Planck Institute for Biology of Ageing, Cologne, Germany.

Marija Kovacevic-Sarmiento (M)

Max Planck Institute for Biology of Ageing, Cologne, Germany.

Constantinos Demetriades (C)

Max Planck Institute for Biology of Ageing, Cologne, Germany. Demetriades@age.mpg.de.
Cologne Graduate School of Ageing Research, Cologne, Germany. Demetriades@age.mpg.de.
Cologne Excellence Cluster on Cellular Stress Responses in Aging-Associated Diseases, University of Cologne, Cologne, Germany. Demetriades@age.mpg.de.

Classifications MeSH