This protein contributes to histone modification by positioning histone H3 for efficient trimethylation at lysine-4 and stimulating histone methyltransferase activities as part of the MLL1/MLL complex and NSL complex, which are essential for epigenetic transcriptional activation. Mutations cause an autosomal dominant neurodevelopmental disorder through loss-of-function mechanisms. The gene is highly intolerant to loss-of-function variants, indicating haploinsufficiency as the primary pathogenic mechanism.

OMIMResearchSummary from RefSeq, UniProt, Mechanism
MultiplemechanismLOEUF 0.12
Clinical SummaryWDR5
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Gene-Disease Validity (ClinGen)
congenital heart disease · ADLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
Some data sources returned errors (1)

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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.12LOEUF
pLI 1.000
Z-score 4.54
OE 0.00 (0.000.12)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
3.39Z-score
OE missense 0.32 (0.260.39)
63 obs / 197.0 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.00 (0.000.12)
00.351.4
Missense OE0.32 (0.260.39)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 0 / 24.0Missense obs/exp: 63 / 197.0Syn Z: 0.26
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
moderateWDR5-related neurodevelopmental disorderOTHERAD
DN
0.4190th %ile
GOF
0.3392th %ile
LOF
0.77top 5%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to loss-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFprediction above median · LOEUF 0.12
DN1 literature citation

Literature Evidence

DNThese results, together with the clustering of variants in a specific region of WDR5 and the absence of truncating variants so far, suggest that dominant-negative or gain-of-function mechanisms might be at play.PMID:36408368

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

WDR5 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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