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10 sources retrieved · Most recent: April 2026 · Index updated 14 days ago
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WDR24
WD repeat domain 24
Chromosome 16 · 16p13.3
NCBI Gene: 84219Ensembl: ENSG00000127580.18HGNC: HGNC:20852UniProt: Q96S15
56PubMed Papers
20Diseases
0Drugs
0Pathogenic Variants
DATA QUALITY
✓ Experimental GO Evidence✓ Swiss-Prot Reviewed
protein localization to plasma membraneprotein bindingubiquitin protein ligase activityregulation of autophagyneurodegenerative diseasesmoking initiationBlackfan-Diamond anemiaprimary familial polycythemia due to EPO receptor mutation
✦AI Summary

WDR24 is the catalytic component of the GATOR2 complex, a nutrient-sensing hub that activates mTORC1 signaling in response to amino acid availability 1. GATOR2 forms a large cage-like structure with two-fold symmetry, where WDR24 contributes catalytic WD40 β-propeller domains that interact with amino acid sensors Sestrin2 (leucine sensor) and CASTOR1 (arginine sensor) 123. Mechanistically, WDR24 mediates Lys-6-linked ubiquitination of NPRL2, a GATOR1 component, thereby inactivating GATOR1 and allowing mTORC1 activation when amino acids are abundant 45. In amino acid-depleted conditions, GATOR2 is inhibited by sensor protein binding, which restricts WDR24's dynamic propeller domain, preventing GATOR1 inhibition and suppressing mTORC1 2. Beyond amino acid sensing, WDR24 regulates glucose-dependent mTORC1 activation through AMPK-mediated phosphorylation at S155 5. WDR24 also promotes lysosomal acidification and autophagic flux 6. Disease relevance includes cancer, where dysregulated WDR24-GATOR2 signaling drives hyperactive mTORC1 7, making this axis a therapeutic target for conditions with aberrant mTORC1 activation.

Sources cited
1
WDR24 is a core component of the GATOR2 complex with cage-like structure containing WD40 β-propeller domains
PMID: 35831510
2
GATOR2 interacts with Sestrin2 leucine sensor to regulate mTORC1
PMID: 26449471
3
GATOR2 negatively regulates GATOR1 to activate mTORC1 signaling and is mutated in cancer
PMID: 23723238
4
WDR24 mediates Lys-6-linked ubiquitination of NPRL2 to inactivate GATOR1
PMID: 36528027
5
AMPK phosphorylates WDR24 at S155 to suppress glucose-mediated mTORC1 activation
PMID: 36732624
6
GATOR2 promotes lysosomal acidification and autophagic flux
PMID: 27166823
7
WDR24 β-propeller is restricted by amino acid sensors to control mTORC1 activation
PMID: 40836086
8
WDR24 interacts with Sestrin2 and contributes to GATOR2 inhibition by amino acid sensors
PMID: 40742811
9
CDK5-PRMT1 pathway methylates WDR24 to promote mTORC1 activation in cancer
PMID: 36995937
Disease Associationsⓘ20
neurodegenerative diseaseOpen Targets
0.37Weak
smoking initiationOpen Targets
0.13Weak
Blackfan-Diamond anemiaOpen Targets
0.05Suggestive
primary familial polycythemia due to EPO receptor mutationOpen Targets
0.05Suggestive
alpha thalassemia-intellectual disability syndrome type 1Open Targets
0.05Suggestive
Alpha-thalassemia - intellectual disability syndrome linked to chromosome 16Open Targets
0.05Suggestive
hemoglobin D diseaseOpen Targets
0.04Suggestive
hemolytic anemia due to diphosphoglycerate mutase deficiencyOpen Targets
0.04Suggestive
delta-beta-thalassemiaOpen Targets
0.04Suggestive
Hemoglobin C - beta-thalassemiaOpen Targets
0.04Suggestive
hemoglobin C-beta-thalassemia syndromeOpen Targets
0.04Suggestive
erythrocytosis, familial, 3Open Targets
0.04Suggestive
erythrocytosis, familial, 6Open Targets
0.04Suggestive
hepatocellular carcinomaOpen Targets
0.04Suggestive
PyropoikilocytosisOpen Targets
0.04Suggestive
pyropoikilocytosis, hereditaryOpen Targets
0.04Suggestive
erythrocytosis, familial, 4Open Targets
0.03Suggestive
risk-taking behaviourOpen Targets
0.03Suggestive
atrial fibrillationOpen Targets
0.03Suggestive
neoplasmOpen Targets
0.02Suggestive
Pathogenic Variants
No pathogenic variants reported on ClinVar for this gene.
View on ClinVar ↗
Related Genes
LAMTOR3Protein interaction100%SESN1Protein interaction100%LAMTOR1Protein interaction100%LAMP1Protein interaction100%NPRL3Protein interaction100%DEPDC5Protein interaction100%
Tissue Expression6 tissues
Liver
100%
Ovary
77%
Brain
70%
Lung
59%
Bone Marrow
35%
Heart
29%
Gene Interaction Network
Click a node to explore
WDR24LAMTOR3SESN1LAMTOR1LAMP1NPRL3DEPDC5
PROTEIN STRUCTURE
Preparing viewer…
PDB9LWF · 3.41 Å · EM
View on RCSB ↗
Constraintⓘ
LOEUFⓘ
0.72LoF Tolerant
pLIⓘ
0.00Tolerant
Observed/Expected LoF0.54 [0.40–0.72]
RankingsWhere WDR24 stands among ~20K protein-coding genes
  • #8,155of 20,598
    Most Researched56
  • #5,540of 17,882
    Most Constrained (LOEUF)0.72
Genes detectedWDR24
Sources retrieved10 papers
Response time—
📄 Sources
10▼
1
Structure of the nutrient-sensing hub GATOR2.
PMID: 35831510
Nature · 2022
1.00
2
Sestrin2 is a leucine sensor for the mTORC1 pathway.
PMID: 26449471
Science · 2016
0.90
3
A Tumor suppressor complex with GAP activity for the Rag GTPases that signal amino acid sufficiency to mTORC1.
PMID: 23723238
Science · 2013
0.80
4
Genome-wide CRISPR screens identify ILF3 as a mediator of mTORC1-dependent amino acid sensing.
PMID: 37037994
Nat Cell Biol · 2023
0.70
5
AMPK-dependent phosphorylation of the GATOR2 component WDR24 suppresses glucose-mediated mTORC1 activation.
PMID: 36732624
Nat Metab · 2023
0.60