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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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DCAF17
DDB1 and CUL4 associated factor 17
Chromosome 2 Β· 2q31.1
NCBI Gene: 80067Ensembl: ENSG00000115827.14HGNC: HGNC:25784UniProt: F5H7W1
35PubMed Papers
21Diseases
0Drugs
57Pathogenic Variants
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
protein bindingCul4-RING E3 ubiquitin ligase complexnucleoplasmspermatogenesisWoodhouse-Sakati syndromeDystonianeurodegeneration with brain iron accumulationneurodegenerative disease
✦AI Summary

DCAF17 (DDB1 and CUL4 associated factor 17) functions as a substrate receptor for the CUL4-DDB1 E3 ubiquitin-protein ligase complex, playing a critical role in protein degradation pathways 1. The protein is essential for gonadal development and fertility, with loss-of-function variants causing severe reproductive dysfunction in both males and females 2. Pathogenic mutations in DCAF17 cause Woodhouse-Sakati syndrome (WSS), a rare autosomal recessive multisystemic disorder characterized by hypogonadism, alopecia, diabetes mellitus, intellectual disability, and sensorineural hearing loss 34. The syndrome shows significant phenotypic heterogeneity, with ectodermal and endocrine manifestations being the most consistent features 3. Various types of mutations have been identified, including splice site variants, frameshift deletions, and nonsense mutations, all leading to loss of protein function 56. DCAF17 variants are also associated with primary ovarian insufficiency and have been implicated in neurodegeneration with brain iron accumulation (NBIA), suggesting broader roles in cellular metabolism and neurological function 78. The protein appears critical for mammalian reproductive development, with mouse models demonstrating male infertility and female subfertility when Dcaf17 function is disrupted 2.

Sources cited
1
DCAF17 functions as a substrate receptor for CUL4-DDB1 E3 ubiquitin ligase complex in protein degradation
PMID: 28302793
2
Loss-of-function DCAF17 variants cause reproductive dysfunction and mouse models show infertility
PMID: 29178422
3
DCAF17 mutations cause Woodhouse-Sakati syndrome with ectodermal and endocrine manifestations
PMID: 34590781
4
WSS is characterized by hypogonadism, alopecia, diabetes, intellectual disability, and hearing loss
PMID: 31472064
5
Various mutation types including splice site variants cause loss of DCAF17 function
PMID: 35876063
6
Splicing mutations result in truncated functionless protein
PMID: 34877714
7
DCAF17 variants are associated with primary ovarian insufficiency
PMID: 34794894
8
DCAF17 mutations are implicated in neurodegeneration with brain iron accumulation
PMID: 39419454
Disease Associationsβ“˜21
Woodhouse-Sakati syndromeOpen Targets
0.78Strong
DystoniaOpen Targets
0.46Moderate
neurodegeneration with brain iron accumulationOpen Targets
0.41Moderate
neurodegenerative diseaseOpen Targets
0.26Weak
osteoarthritis, kneeOpen Targets
0.25Weak
genetic disorderOpen Targets
0.19Weak
hemiplegiaOpen Targets
0.18Weak
azoospermiaOpen Targets
0.10Weak
multiple sclerosisOpen Targets
0.08Suggestive
partial chromosome Y deletionOpen Targets
0.07Suggestive
spermatogenic failure 78Open Targets
0.07Suggestive
pericarditisOpen Targets
0.07Suggestive
spermatogenic failure 65Open Targets
0.07Suggestive
spermatogenic failure 54Open Targets
0.07Suggestive
spermatogenic failure 84Open Targets
0.07Suggestive
spermatogenic failure 93Open Targets
0.07Suggestive
spermatogenic failure 91Open Targets
0.07Suggestive
spermatogenic failure 56Open Targets
0.07Suggestive
spermatogenic failure 92Open Targets
0.07Suggestive
spermatogenic failure 94Open Targets
0.07Suggestive
Woodhouse-Sakati syndromeUniProt
Pathogenic Variants57
NM_025000.4(DCAF17):c.906G>A (p.Trp302Ter)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2025β†’ Residue 302
NM_025000.4(DCAF17):c.1A>G (p.Met1Val)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2025β†’ Residue 1
NM_025000.4(DCAF17):c.127-1G>CPathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2025
NM_025000.4(DCAF17):c.436del (p.Ala147fs)Pathogenic
Woodhouse-Sakati syndrome|not provided|DCAF17-related disorder
β˜…β˜…β˜†β˜†2024β†’ Residue 147
NM_025000.4(DCAF17):c.413del (p.Gly138fs)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2024β†’ Residue 138
NM_025000.4(DCAF17):c.387G>A (p.Trp129Ter)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2024β†’ Residue 129
NM_025000.4(DCAF17):c.1488_1489del (p.Arg496fs)Pathogenic
Woodhouse-Sakati syndrome|Neurodegeneration with brain iron accumulation
β˜…β˜…β˜†β˜†2023β†’ Residue 496
NM_025000.4(DCAF17):c.85C>T (p.Gln29Ter)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2023β†’ Residue 29
NM_025000.4(DCAF17):c.289dup (p.Ile97fs)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜…β˜†β˜†2023β†’ Residue 97
NM_025000.4(DCAF17):c.252_262del (p.Lys84fs)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2025β†’ Residue 84
NM_025000.4(DCAF17):c.1204dup (p.Thr402fs)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2025β†’ Residue 402
NM_025000.4(DCAF17):c.839-2A>GLikely pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2025
NM_025000.4(DCAF17):c.739C>T (p.Gln247Ter)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2025β†’ Residue 247
NM_025000.4(DCAF17):c.1494del (p.Phe498fs)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2025β†’ Residue 498
NM_025000.4(DCAF17):c.1231dup (p.Ser411fs)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2024β†’ Residue 411
NM_025000.4(DCAF17):c.322-2A>GLikely pathogenic
Woodhouse-Sakati syndrome|Papillary renal cell carcinoma type 1
β˜…β˜†β˜†β˜†2024
NM_025000.4(DCAF17):c.628-2A>CLikely pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2024
NM_025000.4(DCAF17):c.733-1G>CLikely pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2024
NM_025000.4(DCAF17):c.308G>A (p.Trp103Ter)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2024β†’ Residue 103
NM_025000.4(DCAF17):c.289del (p.Lys96_Ile97insTer)Pathogenic
Woodhouse-Sakati syndrome
β˜…β˜†β˜†β˜†2024β†’ Residue 96
View on ClinVar β†—
Related Genes
SPMIP7Shared pathway100%CBY3Shared pathway100%ARRDC5Shared pathway100%C3orf62Shared pathway100%TXNDC8Shared pathway100%TDRPShared pathway100%
Tissue Expression6 tissues
Ovary
100%
Bone Marrow
78%
Heart
73%
Brain
68%
Liver
59%
Lung
49%
Gene Interaction Network
Click a node to explore
DCAF17SPMIP7CBY3ARRDC5C3orf62TXNDC8TDRP
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q5H9S7
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.85LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.64 [0.48–0.85]
RankingsWhere DCAF17 stands among ~20K protein-coding genes
  • #10,952of 20,598
    Most Researched35
  • #1,220of 5,498
    Most Pathogenic Variants57 Β· top quartile
  • #7,454of 17,882
    Most Constrained (LOEUF)0.85
Genes detectedDCAF17
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Genetics of ovarian insufficiency and defects of folliculogenesis.
PMID: 34794894
Best Pract Res Clin Endocrinol Metab Β· 2022
1.00
2
Anticancer sulfonamides target splicing by inducing RBM39 degradation via recruitment to DCAF15.
PMID: 28302793
Science Β· 2017
0.90
3
Novel inactivating mutations of the DCAF17 gene in American and Turkish families cause male infertility and female subfertility in the mouse model.
PMID: 29178422
Clin Genet Β· 2018
0.80
4
Expanding on the phenotypic spectrum of Woodhouse-Sakati syndrome due to founder pathogenic variant in DCAF17: Report of 58 additional patients from Qatar and literature review.
PMID: 34590781
Am J Med Genet A Β· 2022
0.70
5
Whole exome sequencing and transcript analysis discover a novel pathogenic splice site mutation in DCAF17 gene underlying Woodhouse-Sakati syndrome.
PMID: 35876063
J Neuroendocrinol Β· 2022
0.60