WDFY1

Chr 2

WD repeat and FYVE domain containing 1

Also known as: FENS-1, FENS1, WDF1, ZFYVE17

WDFY1 encodes a phosphatidylinositol 3-phosphate binding protein that localizes to early endosomes and positively regulates TLR3- and TLR4-mediated signaling pathways, promoting inflammatory cytokine production. The gene is highly constrained against loss-of-function variants (LOEUF 0.752), but specific disease associations have not been established in the provided data. Additional clinical and genetic evidence would be needed to determine the phenotypic consequences of WDFY1 mutations.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
8
Pubs (1 yr)
32
P/LP submissions
0%
P/LP missense
0.75
LOEUF
Mechanism
Clinical SummaryWDFY1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
32 unique Pathogenic / Likely Pathogenic· 64 VUS of 121 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.75LOEUF
pLI 0.000
Z-score 2.49
OE 0.45 (0.280.75)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.43Z-score
OE missense 0.75 (0.660.84)
190 obs / 254.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.45 (0.280.75)
00.351.4
Missense OE0.75 (0.660.84)
00.61.4
Synonymous OE0.89
01.21.6
LoF obs/exp: 11 / 24.2Missense obs/exp: 190 / 254.3Syn Z: 0.87

ClinVar Variant Classifications

121 submitted variants in ClinVar

Classification Summary

Pathogenic31
Likely Pathogenic1
VUS64
31
Pathogenic
1
Likely Pathogenic
64
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
31
0
31
Likely Pathogenic
0
0
1
0
1
VUS
0
62
2
0
64
Likely Benign
0
0
0
0
0
Benign
0
0
0
0
0
Total06234096

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

WDFY1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 2 results · since 2015Search PubMed ↗