Affinage

WDR24

GATOR2 complex protein WDR24 · UniProt Q96S15

Length
790 aa
Mass
88.2 kDa
Annotated
2026-06-11
19 papers in source corpus 12 papers cited in narrative 12 extracted findings
Cross-family judge vs UniProt: Affinage preferred faithfulness: 6/7 claims corpus-supported (86%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

WDR24 is a core scaffold subunit of the GATOR2 complex that acts as a positive regulator of amino acid signaling to mTORC1 upstream of the Rag GTPases, where GATOR2 negatively regulates the GATOR1 GAP complex (DEPDC5/NPRL2/NPRL3) toward RagA/RagB (PMID:23723238). Within the ~1.1 MDa two-fold symmetric, cage-like GATOR2 architecture, WDR24 contributes WD40 β-propeller dimers to an octagonal scaffold built from WDR24, MIOS, and WDR59 circularized through non-catalytic RING domains and α-solenoids, with SEH1L and SEC13 stabilizing the assembly by β-propeller blade donation (PMID:35831510). The WDR24 β-propeller is the docking site for the leucine sensor Sestrin2, engaging it via the WDR24-SEH1L subcomplex; sensor binding restricts the dynamic WDR24 propeller that is essential for nutrient-dependent mTORC1 activation, and amino-acid-induced conformational changes drive sensor dissociation to relieve this restraint (PMID:40836086, PMID:40742811). WDR24 is also the GATOR2 component required for the Sestrin2-GATOR2 interaction that couples to mTORC2 through WDR59 to activate AKT (PMID:31915252), and lysosomal localization of GATOR2 imparted through WDR24 is sufficient for amino-acid-dependent mTORC1 signaling (PMID:37037994). The complex serves as a signal convergence point regulated by post-translational modification: PRMT1-mediated arginine methylation activates the WDR24/mTORC1 axis in response to amino acids (PMID:36995937), whereas AMPK directly phosphorylates WDR24 at Ser155 upon glucose deprivation to disrupt GATOR2 integrity and suppress mTORC1, a regulatory event essential in vivo as phosphomimetic knock-in mice are embryonic-lethal (PMID:36732624). WDR24 protein stability within the complex depends on WDR59, which protects it from proteasomal degradation (PMID:36577058). Beyond mTORC1, WDR24 has a TORC1-independent role in lysosome acidification and autophagic flux (PMID:27166823).

Mechanistic history

Synthesis pass · year-by-year structured walk · 11 steps
  1. 2013 High

    Established WDR24's foundational identity as a GATOR2 subunit and placed it in the amino-acid-sensing hierarchy upstream of mTORC1, defining the double-negative GATOR2→GATOR1→Rag logic.

    Evidence RNAi knockdown, reciprocal Co-IP, and epistasis with phospho-S6K1 mTORC1 readouts

    PMID:23723238

    Open questions at the time
    • Mechanism by which GATOR2 represses GATOR1 not resolved
    • Structural basis of subunit assembly unknown at this stage
    • How amino acid signals are received not addressed
  2. 2016 High

    Demonstrated that WDR24 is required for TORC1 activation in vivo while also carrying a TORC1-independent function in lysosome dynamics, separating its growth-signaling and organelle roles.

    Evidence Drosophila null allele characterization, genetic epistasis with GATOR1, and HeLa knockout cell lysosome/autophagy assays

    PMID:27166823

    Open questions at the time
    • Molecular basis of the TORC1-independent lysosomal function unknown
    • Direct effector of lysosome acidification not identified
  3. 2020 High

    Identified WDR24 as the obligate bridge linking the leucine sensor Sestrin2 to GATOR2 and, via WDR59, to mTORC2/AKT, extending GATOR2 signaling beyond mTORC1.

    Evidence Co-IP, siRNA knockdown, WDR24 deletion, and in vitro kinase/AKT activation assays

    PMID:31915252

    Open questions at the time
    • Structural interface of Sestrin2-WDR24 not defined here
    • Direct vs scaffold role of WDR24 in mTORC2 activation unclear
  4. 2022 High

    Resolved the GATOR2 cage architecture, showing WDR24 contributes WD40 β-propeller dimers that form the interaction platform for SESN2, CASTOR1, and GATOR1.

    Evidence Cryo-EM structure with biochemical reconstitution

    PMID:35831510

    Open questions at the time
    • Static structure does not capture sensor-induced conformational dynamics
    • Mechanism of GATOR1 regulation by GATOR2 still structurally unresolved
  5. 2022 Medium

    Showed WDR24 protein abundance within GATOR2 is maintained by WDR59, which shields it from proteasomal turnover, linking complex assembly to subunit stability and TORC1 output.

    Evidence WDR59 knockout HeLa cells with proteasome-inhibitor rescue and WDR24 immunoblot

    PMID:36577058

    Open questions at the time
    • E3 ligase targeting WDR24 not identified
    • Single lab, single cell system
  6. 2023 High

    Defined AMPK as a direct glucose-sensitive negative regulator that phosphorylates WDR24 Ser155 to disassemble GATOR2 and suppress mTORC1, validated by knock-in mouse phenotypes.

    Evidence In vitro kinase assay with site-specific mutagenesis and S155D/S155A knock-in mice with mTORC1 and fasting phenotypes

    PMID:36732624

    Open questions at the time
    • Structural consequence of Ser155 phosphorylation on the cage not visualized
    • How disassembly is reversed upon glucose restoration unknown
  7. 2023 Medium

    Identified arginine methylation of WDR24 by PRMT1, driven by a CDK5-PRMT1 amino-acid-responsive axis, as an activating modification promoting mTORC1 and tumor growth.

    Evidence Co-IP, arginine methylation assays, subcellular fractionation, knockdown, and HCC xenograft models

    PMID:36995937

    Open questions at the time
    • Specific WDR24 methylation site not mapped
    • Mechanistic link between methylation and complex activation undefined
  8. 2023 Medium

    Established that lysosomal recruitment of GATOR2, mediated through WDR24, is the rate-limiting step for amino-acid-dependent mTORC1, with ILF3 as the tether.

    Evidence Genome-wide CRISPR screen and lysosome-targeting WDR24 construct rescue with pS6 FACS

    PMID:37037994

    Open questions at the time
    • Direct WDR24-ILF3 contact not structurally defined
    • Regulation of the tethering step by nutrients unclear
  9. 2024 Medium

    Revealed WDR24 as a target of pathogen-derived metabolite signaling, where M. tuberculosis D-serine binds WDR24 to suppress mTORC1 in CD8+ T cells and dampen IFN-γ.

    Evidence Metabolomic/proteomic/genetic approaches with direct D-serine–WDR24 interaction assay and mTORC1 readouts in CD8+ T cells

    PMID:38806671

    Open questions at the time
    • Binding site of D-serine on WDR24 not mapped
    • Structural mechanism of inhibition unknown
    • Single lab
  10. 2025 High

    Cryo-EM and HDX-MS of sensor-bound GATOR2 pinpointed the WDR24-SEH1L subcomplex as the Sestrin2 interface and showed sensor binding restricts the dynamic WDR24 β-propeller required for activation.

    Evidence Cryo-EM structures in apo and inhibitory states, interface mutagenesis, and HDX-MS with functional mTORC1 assays

    PMID:40742811 PMID:40836086

    Open questions at the time
    • Coupling of WDR24 propeller dynamics to downstream GATOR1 regulation not fully resolved
    • Active (sensor-free) signaling conformation not captured
  11. 2025 Medium

    Extended WDR24 signaling into the DNA damage response, showing radiation-induced AMPK phosphorylation of WDR24 drives NPRL2 release and nuclear translocation to enhance DNA repair.

    Evidence Co-IP, nuclear fractionation, AMPK inhibition, and radiosensitization assays in CRC cells in vitro and in vivo

    PMID:41584340

    Open questions at the time
    • Phosphorylation site mediating this effect not specified
    • Direct WDR24 contribution vs upstream GATOR2 disassembly not separated
    • Single study

Open questions

Synthesis pass · forward-looking unresolved questions
  • How the multiple WDR24 post-translational and metabolite inputs (Ser155 phosphorylation, PRMT1 methylation, D-serine binding) are structurally integrated on the GATOR2 cage to set a graded mTORC1 output remains unresolved.
  • No structure of a modified WDR24 within GATOR2
  • Quantitative hierarchy among amino acid, glucose, and metabolite signals undefined
  • Mechanism converting WDR24 propeller dynamics into GATOR1 inhibition unknown

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0060090 molecular adaptor activity 3 GO:0098772 molecular function regulator activity 2 GO:0140299 molecular sensor activity 2
Localization
GO:0005764 lysosome 2 GO:0005829 cytosol 1
Pathway
R-HSA-162582 Signal Transduction 2 R-HSA-8953897 Cellular responses to stimuli 2 R-HSA-9612973 Autophagy 1
Complex memberships
GATOR2

Evidence

Reading pass · 12 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2013 WDR24 is a subunit of the GATOR2 complex (along with Mios, WDR59, Seh1L, and Sec13), which positively regulates mTORC1 signaling upstream of the Rag GTPases. Inhibition of WDR24/GATOR2 suppresses mTORC1 signaling, and epistasis analysis shows GATOR2 negatively regulates GATOR1 (DEPDC5/Nprl2/Nprl3), which itself has GAP activity toward RagA/RagB. RNAi knockdown, epistasis analysis, Co-immunoprecipitation, mTORC1 activity assays (phospho-S6K1) Science High 23723238
2022 Cryo-EM structure of human GATOR2 revealed a ~1.1 MDa two-fold symmetric cage-like architecture with an octagonal scaffold containing two WDR24, four MIOS, and two WDR59 subunits circularized via non-catalytic RING domains and α-solenoids. WDR24 contributes WD40 β-propeller dimers that mediate interactions with SESN2, CASTOR1, and GATOR1. SEH1L and SEC13 stabilize the complex via β-propeller blade donation. Cryo-electron microscopy, biochemical reconstitution Nature High 35831510
2016 Drosophila Wdr24 (ortholog of WDR24) is required for robust TORC1 activation and cellular growth in vivo. Additionally, Wdr24 has a TORC1-independent function in regulating lysosome dynamics and autophagic flux. In wdr24-/- knockout HeLa cells, Wdr24 promotes lysosome acidification and autophagic flux. Null allele characterization, genetic epistasis with GATOR1 components, biochemical fractionation, cell biological assays, HeLa knockout cell line PLoS genetics High 27166823
2020 WDR24 is the GATOR2 component essential for the Sestrin2-GATOR2 interaction. Deleting or silencing WDR24 completely ablates Sestrin2-induced AKT activation. WDR24 bridges Sestrin2 to the GATOR2 complex, which in turn interacts with mTORC2 through WDR59 to induce AKT activation. Co-immunoprecipitation, siRNA knockdown, WDR24 deletion, in vitro kinase assay, AKT activation assays The Journal of biological chemistry High 31915252
2023 AMPK directly phosphorylates WDR24 at Ser155 upon glucose deprivation, disrupting the integrity of the GATOR2 complex and suppressing mTORC1 activation. Phosphomimetic Wdr24-S155D knock-in mice exhibit early embryonic lethality and reduced mTORC1 activity, while phospho-deficient Wdr24-S155A knock-in mice are more resistant to fasting and display elevated mTORC1 activity. In vitro kinase assay, phosphomimetic and phospho-deficient knock-in mice, Co-immunoprecipitation, mTORC1 activity assays Nature metabolism High 36732624
2023 PRMT1, activated by CDK5-mediated phosphorylation at S307 in response to amino acids, translocates from nucleus to cytoplasm/lysosome and methylates WDR24, an essential GATOR2 component, to activate the mTORC1 pathway. Disruption of the CDK5-PRMT1-WDR24 axis suppresses mTORC1 signaling, HCC cell proliferation, and xenograft tumor growth. Co-immunoprecipitation, arginine methylation assays, subcellular fractionation/localization, siRNA knockdown, xenograft tumor models Cell reports Medium 36995937
2023 ILF3 tethers the GATOR complexes (including WDR24-containing GATOR2) to lysosomes to control mTORC1. Adding a lysosome-targeting sequence to WDR24 directly bypasses the requirement for ILF3 in amino-acid-dependent mTORC1 signalling, demonstrating that lysosomal localization of GATOR2 via WDR24 is functionally sufficient. Genome-wide CRISPR/Cas9 screen, lysosome-targeting sequence rescue experiment, mTORC1 activity assays (pS6 FACS) Nature cell biology Medium 37037994
2025 Cryo-EM structures of GATOR2 bound to Sestrin2 or CASTOR1 revealed that Sestrin2 interacts with the WDR24-Seh1L subcomplex, inducing conformational changes, while CASTOR1 engages Mios WD40 β-propellers. Binding of either sensor restricts the dynamic WDR24 β-propeller, a domain essential for nutrient-dependent mTORC1 activation. Amino acid binding triggers structural rearrangements in sensors that cause their dissociation from GATOR2. Cryo-electron microscopy, site-directed mutagenesis of binding interfaces, functional mTORC1 signaling assays Nature High 40836086
2025 Cryo-EM structures of GATOR2 in three inhibitory states (CASTOR1-bound, Sestrin2-bound, dual-bound) confirmed that Sestrin2 interacts with the WDR24-Seh1L subcomplex while CASTOR1 engages Mios WD40 β-propellers. HDX-MS revealed dynamic motions in apo-GATOR2 and its sensor complexes. Cryo-electron microscopy, hydrogen-deuterium exchange mass spectrometry (HDX-MS) Cell reports High 40742811
2024 D-serine produced by Mycobacterium tuberculosis interacts with WDR24 and inhibits mTORC1 activation in CD8+ T cells, decreasing T-bet expression and reducing IFN-γ production. Metabolomic, proteomic and genetic approaches; direct interaction assay between D-serine and WDR24; mTORC1 activity assays in CD8+ T cells Nature microbiology Medium 38806671
2022 In mammalian HeLa cells, Wdr59 (another GATOR2 component) prevents the proteolytic destruction of GATOR2 proteins Mio and Wdr24. In Wdr59 knockout HeLa cells, reduced Wdr24 protein levels correlated with reduced TORC1 activity, which was restored along with Wdr24 levels upon proteasome inhibition. Wdr59 knockout HeLa cells, proteasome inhibition rescue, immunoblot for Wdr24 protein levels, mTORC1 activity assays Proceedings of the National Academy of Sciences of the United States of America Medium 36577058
2025 Radiation-induced AMPK activation phosphorylates WDR24, which promotes NPRL2 dissociation from the GATOR1 complex and facilitates NPRL2 nuclear translocation. Nuclear NPRL2 then inactivates E3 ubiquitin ligases HERC2 and RNF8 to enhance DNA repair. AMPK inhibition blocks this cascade, impairing DNA repair and radiosensitizing CRC cells. Co-immunoprecipitation, nuclear fractionation, AMPK inhibition, in vitro and in vivo radiosensitization assays Acta pharmaceutica Sinica. B Medium 41584340

Source papers

Stage 0 corpus · 19 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2013 A Tumor suppressor complex with GAP activity for the Rag GTPases that signal amino acid sufficiency to mTORC1. Science (New York, N.Y.) 888 23723238
2016 Involvement of GATOR complex genes in familial focal epilepsies and focal cortical dysplasia. Epilepsia 140 27173016
2022 Structure of the nutrient-sensing hub GATOR2. Nature 71 35831510
2022 Dysregulated Amino Acid Sensing Drives Colorectal Cancer Growth and Metabolic Reprogramming Leading to Chemoresistance. Gastroenterology 62 36410445
2020 The GATOR2-mTORC2 axis mediates Sestrin2-induced AKT Ser/Thr kinase activation. The Journal of biological chemistry 52 31915252
2016 The GATOR2 Component Wdr24 Regulates TORC1 Activity and Lysosome Function. PLoS genetics 45 27166823
2023 AMPK-dependent phosphorylation of the GATOR2 component WDR24 suppresses glucose-mediated mTORC1 activation. Nature metabolism 38 36732624
2023 Genome-wide CRISPR screens identify ILF3 as a mediator of mTORC1-dependent amino acid sensing. Nature cell biology 30 37037994
2019 Pre-clinical activity of combined LSD1 and mTORC1 inhibition in MLL-translocated acute myeloid leukaemia. Leukemia 29 31780813
2024 Mycobacterium tuberculosis produces D-serine under hypoxia to limit CD8+ T cell-dependent immunity in mice. Nature microbiology 28 38806671
2023 CDK5-PRMT1-WDR24 signaling cascade promotes mTORC1 signaling and tumor growth. Cell reports 28 36995937
2025 Structural basis for the dynamic regulation of mTORC1 by amino acids. Nature 11 40836086
2025 Cryo-EM structures of amino acid sensors bound to the human GATOR2 complex. Cell reports 9 40742811
2024 New insights into GATOR2-dependent interactions and its conformational changes in amino acid sensing. Bioscience reports 7 38372438
2022 Wdr59 promotes or inhibits TORC1 activity depending on cellular context. Proceedings of the National Academy of Sciences of the United States of America 7 36577058
2023 Identification of Epigenetically Regulated Genes Distinguishing Intracranial from Extracranial Melanoma Metastases. The Journal of investigative dermatology 4 36716920
2024 Identifying key pathogenic mechanisms and potential intervention targets for recurrence after laryngeal cancer treatment through bioinformatics screening. Translational cancer research 1 39145096
2025 Radiation-induced nuclear translocation of NPRL2 hijacks E3 ubiquitin ligases to enhance DNA repair via the AMPK/WDR24 axis, contributing to CRC radioresistance. Acta pharmaceutica Sinica. B 0 41584340
2023 The Challenge of Somatic Variants in Focal Cortical Dysplasia. Innovations in clinical neuroscience 0 38193103

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