WDR41 Gene

HGNC Family	WD repeat domain containing (WDR)
Name	WD repeat domain 41
Description	Enables GTPase activator activity. Contributes to guanyl-nucleotide exchange factor activity. Involved in regulation of autophagy. Located in cytoplasm. Part of guanyl-nucleotide exchange factor complex. [provided by Alliance of Genome Resources, Mar 2025]
Summary	{"type": "root", "children": [{"type": "p", "children": [{"type": "t", "text": "\nWDR41 is a multifunctional WD‐repeat protein that plays a critical role as part of a larger protein complex with C9orf72 and SMCR8. In this assembly, WDR41 interacts indirectly with C9orf72 by binding to the DENN domain of SMCR8, thereby contributing to the complex’s dimerization and GTPase activating protein (GAP) functions. The complex regulates early autophagic events and lysosome homeostasis, as evidenced by its association with the FIP200/Ulk1 complex and its recruitment to lysosomes under amino acid deprivation. Structural studies further reveal that WDR41 not only helps mediate the interactions necessary for the proper assembly of the C9orf72-SMCR8 complex but also, through a specific association with the lysosomal transporter PQLC2, facilitates a nutrient-dependent recruitment mechanism that is pivotal for mTORC1 signaling."}, {"type": "fg", "children": [{"type": "fg_fs", "start_ref": "1", "end_ref": "5"}]}, {"type": "t", "text": "\n"}]}, {"type": "t", "text": "\n\n"}, {"type": "p", "children": [{"type": "t", "text": "\nBeyond its established role in autophagy and lysosome dynamics, evidence from genetic association studies indicates that WDR41 may also contribute to neurodevelopment. Variations in and around the WDR41 gene have been linked to differences in caudate volume, suggesting a potential involvement in dopamine signaling and the regulation of brain structure during development."}, {"type": "fg", "children": [{"type": "fg_f", "ref": "6"}]}, {"type": "t", "text": "\n"}]}, {"type": "t", "text": "\n\n"}, {"type": "p", "children": [{"type": "t", "text": "\nIn addition, aberrant regulation of WDR41 appears to have implications in tumorigenesis. In the context of breast cancer—especially triple‐negative breast cancer—hypermethylation and down‐regulation of WDR41 correlate with enhanced oncogenic behaviors, including increased cell viability, migration, and dysregulated cell cycle progression. Mechanistic studies have further demonstrated that reduced levels of WDR41 are associated with the activation of the AKT/GSK-3β/β-catenin pathway, whereas re-establishing WDR41 expression can suppress these tumor-promoting signals and restrain tumor growth."}, {"type": "fg", "children": [{"type": "fg_f", "ref": "7"}]}, {"type": "t", "text": "\n"}]}, {"type": "rg", "children": [{"type": "r", "ref": 1, "children": [{"type": "t", "text": "Peter M Sullivan, Xiaolai Zhou, Adam M Robins, et al. "}, {"type": "b", "children": [{"type": "t", "text": "The ALS/FTLD associated protein C9orf72 associates with SMCR8 and WDR41 to regulate the autophagy-lysosome pathway."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "Acta Neuropathol Commun (2016)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1186/s40478-016-0324-5"}], "href": "https://doi.org/10.1186/s40478-016-0324-5"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "27193190"}], "href": "https://pubmed.ncbi.nlm.nih.gov/27193190"}]}, {"type": "r", "ref": 2, "children": [{"type": "t", "text": "Dan Tang, Jingwen Sheng, Liangting Xu, et al. "}, {"type": "b", "children": [{"type": "t", "text": "Cryo-EM structure of C9ORF72-SMCR8-WDR41 reveals the role as a GAP for Rab8a and Rab11a."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "Proc Natl Acad Sci U S A (2020)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1073/pnas.2002110117"}], "href": "https://doi.org/10.1073/pnas.2002110117"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "32303654"}], "href": "https://pubmed.ncbi.nlm.nih.gov/32303654"}]}, {"type": "r", "ref": 3, "children": [{"type": "t", "text": "Ming-Yuan Su, Simon A Fromm, Roberto Zoncu, et al. "}, {"type": "b", "children": [{"type": "t", "text": "Structure of the C9orf72 ARF GAP complex that is haploinsufficient in ALS and FTD."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "Nature (2020)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1038/s41586-020-2633-x"}], "href": "https://doi.org/10.1038/s41586-020-2633-x"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "32848248"}], "href": "https://pubmed.ncbi.nlm.nih.gov/32848248"}]}, {"type": "r", "ref": 4, "children": [{"type": "t", "text": "Joseph Amick, Arun Kumar Tharkeshwar, Catherine Amaya, et al. "}, {"type": "b", "children": [{"type": "t", "text": "WDR41 supports lysosomal response to changes in amino acid availability."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "Mol Biol Cell (2018)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1091/mbc.E17-12-0703"}], "href": "https://doi.org/10.1091/mbc.E17-12-0703"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "29995611"}], "href": "https://pubmed.ncbi.nlm.nih.gov/29995611"}]}, {"type": "r", "ref": 5, "children": [{"type": "t", "text": "Gabriel Talaia, Joseph Amick, Shawn M Ferguson "}, {"type": "b", "children": [{"type": "t", "text": "Receptor-like role for PQLC2 amino acid transporter in the lysosomal sensing of cationic amino acids."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "Proc Natl Acad Sci U S A (2021)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1073/pnas.2014941118"}], "href": "https://doi.org/10.1073/pnas.2014941118"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "33597295"}], "href": "https://pubmed.ncbi.nlm.nih.gov/33597295"}]}, {"type": "r", "ref": 6, "children": [{"type": "t", "text": "J L Stein, D P Hibar, S K Madsen, et al. "}, {"type": "b", "children": [{"type": "t", "text": "Discovery and replication of dopamine-related gene effects on caudate volume in young and elderly populations (N=1198) using genome-wide search."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "Mol Psychiatry (2011)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1038/mp.2011.32"}], "href": "https://doi.org/10.1038/mp.2011.32"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "21502949"}], "href": "https://pubmed.ncbi.nlm.nih.gov/21502949"}]}, {"type": "r", "ref": 7, "children": [{"type": "t", "text": "Han Wang, Dan Wu, Liangliang Cai, et al. "}, {"type": "b", "children": [{"type": "t", "text": "Aberrant methylation of WD-repeat protein 41 contributes to tumour progression in triple-negative breast cancer."}]}, {"type": "t", "text": " "}, {"type": "i", "children": [{"type": "t", "text": "J Cell Mol Med (2020)"}]}, {"type": "t", "text": " DOI: "}, {"type": "a", "children": [{"type": "t", "text": "10.1111/jcmm.15344"}], "href": "https://doi.org/10.1111/jcmm.15344"}, {"type": "t", "text": " PMID: "}, {"type": "a", "children": [{"type": "t", "text": "32394588"}], "href": "https://pubmed.ncbi.nlm.nih.gov/32394588"}]}]}]}
Synonyms	MSTP048
Proteins	WDR41_HUMAN
NCBI Gene ID	55255
API
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Predicted Functions
Co-expressed Genes
Expression in Tissues and Cell Lines

Functional Associations

WDR41 has 5,704 functional associations with biological entities spanning 9 categories (molecular profile, organism, chemical, functional term, phrase or reference, disease, phenotype or trait, structural feature, cell line, cell type or tissue, gene, protein or microRNA, sequence feature) extracted from 111 datasets.

Click the + buttons to view associations for WDR41 from the datasets below.

If available, associations are ranked by standardized value

Harmonizome 3.0

WDR41 Gene

Functional Associations

Please acknowledge the Harmonizome in your publications by citing the following reference(s):

	Dataset	Summary
	Allen Brain Atlas Adult Human Brain Tissue Gene Expression Profiles	tissues with high or low expression of WDR41 gene relative to other tissues from the Allen Brain Atlas Adult Human Brain Tissue Gene Expression Profiles dataset.
	Allen Brain Atlas Adult Mouse Brain Tissue Gene Expression Profiles	tissues with high or low expression of WDR41 gene relative to other tissues from the Allen Brain Atlas Adult Mouse Brain Tissue Gene Expression Profiles dataset.
	Allen Brain Atlas Aging Dementia and Traumatic Brain Injury Tissue Sample Gene Expression Profiles	tissue samples with high or low expression of WDR41 gene relative to other tissue samples from the Allen Brain Atlas Aging Dementia and Traumatic Brain Injury Tissue Sample Gene Expression Profiles dataset.
	Allen Brain Atlas Developing Human Brain Tissue Gene Expression Profiles by Microarray	tissue samples with high or low expression of WDR41 gene relative to other tissue samples from the Allen Brain Atlas Developing Human Brain Tissue Gene Expression Profiles by Microarray dataset.
	Allen Brain Atlas Developing Human Brain Tissue Gene Expression Profiles by RNA-seq	tissue samples with high or low expression of WDR41 gene relative to other tissue samples from the Allen Brain Atlas Developing Human Brain Tissue Gene Expression Profiles by RNA-seq dataset.
	Allen Brain Atlas Prenatal Human Brain Tissue Gene Expression Profiles	tissues with high or low expression of WDR41 gene relative to other tissues from the Allen Brain Atlas Prenatal Human Brain Tissue Gene Expression Profiles dataset.
	BioGPS Cell Line Gene Expression Profiles	cell lines with high or low expression of WDR41 gene relative to other cell lines from the BioGPS Cell Line Gene Expression Profiles dataset.
	BioGPS Human Cell Type and Tissue Gene Expression Profiles	cell types and tissues with high or low expression of WDR41 gene relative to other cell types and tissues from the BioGPS Human Cell Type and Tissue Gene Expression Profiles dataset.
	BioGPS Mouse Cell Type and Tissue Gene Expression Profiles	cell types and tissues with high or low expression of WDR41 gene relative to other cell types and tissues from the BioGPS Mouse Cell Type and Tissue Gene Expression Profiles dataset.
	Carcinogenome Chemical Perturbation Carcinogenicity Signatures	small molecule perturbations changing expression of WDR41 gene from the Carcinogenome Chemical Perturbation Carcinogenicity Signatures dataset.
	CCLE Cell Line Gene CNV Profiles	cell lines with high or low copy number of WDR41 gene relative to other cell lines from the CCLE Cell Line Gene CNV Profiles dataset.
	CCLE Cell Line Gene Expression Profiles	cell lines with high or low expression of WDR41 gene relative to other cell lines from the CCLE Cell Line Gene Expression Profiles dataset.
	CCLE Cell Line Proteomics	Cell lines associated with WDR41 protein from the CCLE Cell Line Proteomics dataset.
	CellMarker Gene-Cell Type Associations	cell types associated with WDR41 gene from the CellMarker Gene-Cell Type Associations dataset.
	ChEA Transcription Factor Binding Site Profiles	transcription factor binding site profiles with transcription factor binding evidence at the promoter of WDR41 gene from the CHEA Transcription Factor Binding Site Profiles dataset.
	ChEA Transcription Factor Targets	transcription factors binding the promoter of WDR41 gene in low- or high-throughput transcription factor functional studies from the CHEA Transcription Factor Targets dataset.
	ChEA Transcription Factor Targets 2022	transcription factors binding the promoter of WDR41 gene in low- or high-throughput transcription factor functional studies from the CHEA Transcription Factor Targets 2022 dataset.
	CMAP Signatures of Differentially Expressed Genes for Small Molecules	small molecule perturbations changing expression of WDR41 gene from the CMAP Signatures of Differentially Expressed Genes for Small Molecules dataset.
	COMPARTMENTS Curated Protein Localization Evidence Scores	cellular components containing WDR41 protein from the COMPARTMENTS Curated Protein Localization Evidence Scores dataset.
	COMPARTMENTS Curated Protein Localization Evidence Scores 2025	cellular components containing WDR41 protein from the COMPARTMENTS Curated Protein Localization Evidence Scores 2025 dataset.
	COMPARTMENTS Experimental Protein Localization Evidence Scores	cellular components containing WDR41 protein in low- or high-throughput protein localization assays from the COMPARTMENTS Experimental Protein Localization Evidence Scores dataset.
	COMPARTMENTS Experimental Protein Localization Evidence Scores 2025	cellular components containing WDR41 protein in low- or high-throughput protein localization assays from the COMPARTMENTS Experimental Protein Localization Evidence Scores 2025 dataset.
	COMPARTMENTS Text-mining Protein Localization Evidence Scores 2025	cellular components co-occuring with WDR41 protein in abstracts of biomedical publications from the COMPARTMENTS Text-mining Protein Localization Evidence Scores 2025 dataset.
	COSMIC Cell Line Gene CNV Profiles	cell lines with high or low copy number of WDR41 gene relative to other cell lines from the COSMIC Cell Line Gene CNV Profiles dataset.
	COSMIC Cell Line Gene Mutation Profiles	cell lines with WDR41 gene mutations from the COSMIC Cell Line Gene Mutation Profiles dataset.
	CTD Gene-Chemical Interactions	chemicals interacting with WDR41 gene/protein from the curated CTD Gene-Chemical Interactions dataset.
	CTD Gene-Disease Associations	diseases associated with WDR41 gene/protein from the curated CTD Gene-Disease Associations dataset.
	dbGAP Gene-Trait Associations	traits associated with WDR41 gene in GWAS and other genetic association datasets from the dbGAP Gene-Trait Associations dataset.
	DeepCoverMOA Drug Mechanisms of Action	small molecule perturbations with high or low expression of WDR41 protein relative to other small molecule perturbations from the DeepCoverMOA Drug Mechanisms of Action dataset.
	DepMap CRISPR Gene Dependency	cell lines with fitness changed by WDR41 gene knockdown relative to other cell lines from the DepMap CRISPR Gene Dependency dataset.
	DISEASES Experimental Gene-Disease Association Evidence Scores	diseases associated with WDR41 gene in GWAS datasets from the DISEASES Experimental Gene-Disease Assocation Evidence Scores dataset.
	DISEASES Text-mining Gene-Disease Association Evidence Scores 2025	diseases co-occuring with WDR41 gene in abstracts of biomedical publications from the DISEASES Text-mining Gene-Disease Assocation Evidence Scores 2025 dataset.
	DisGeNET Gene-Disease Associations	diseases associated with WDR41 gene in GWAS and other genetic association datasets from the DisGeNET Gene-Disease Associations dataset.
	DisGeNET Gene-Phenotype Associations	phenotypes associated with WDR41 gene in GWAS and other genetic association datasets from the DisGeNET Gene-Phenoptype Associations dataset.
	ENCODE Histone Modification Site Profiles	histone modification site profiles with high histone modification abundance at WDR41 gene from the ENCODE Histone Modification Site Profiles dataset.
	ENCODE Transcription Factor Binding Site Profiles	transcription factor binding site profiles with transcription factor binding evidence at the promoter of WDR41 gene from the ENCODE Transcription Factor Binding Site Profiles dataset.
	ENCODE Transcription Factor Targets	transcription factors binding the promoter of WDR41 gene in ChIP-seq datasets from the ENCODE Transcription Factor Targets dataset.
	ESCAPE Omics Signatures of Genes and Proteins for Stem Cells	PubMedIDs of publications reporting gene signatures containing WDR41 from the ESCAPE Omics Signatures of Genes and Proteins for Stem Cells dataset.
	GAD Gene-Disease Associations	diseases associated with WDR41 gene in GWAS and other genetic association datasets from the GAD Gene-Disease Associations dataset.
	GDSC Cell Line Gene Expression Profiles	cell lines with high or low expression of WDR41 gene relative to other cell lines from the GDSC Cell Line Gene Expression Profiles dataset.