IRAK1

Chr X

interleukin 1 receptor associated kinase 1

Also known as: IRAK, pelle

The protein functions as a serine/threonine kinase that initiates innate immune responses by mediating Toll-like receptor and IL-1 receptor signaling pathways, leading to NF-kappa B activation and interferon regulatory factor 7 phosphorylation. Mutations cause X-linked immunodeficiency, which typically presents with recurrent infections and increased susceptibility to pyogenic bacteria. This gene is highly constrained against loss-of-function variants and follows X-linked inheritance.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.28
Clinical SummaryIRAK1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.99). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
105 unique Pathogenic / Likely Pathogenic· 75 VUS of 300 total submissions
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.28LOEUF
pLI 0.988
Z-score 3.96
OE 0.09 (0.040.28)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
1.99Z-score
OE missense 0.67 (0.590.75)
189 obs / 283.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.09 (0.040.28)
00.351.4
Missense OE0.67 (0.590.75)
00.61.4
Synonymous OE0.93
01.21.6
LoF obs/exp: 2 / 22.1Missense obs/exp: 189 / 283.2Syn Z: 0.63
DN
0.4090th %ile
GOF
0.6832th %ile
LOF
0.67top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFprediction above median · LOEUF 0.28
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

300 submitted variants in ClinVar

Classification Summary

Pathogenic101
Likely Pathogenic4
VUS75
Likely Benign20
Benign11
Conflicting2
101
Pathogenic
4
Likely Pathogenic
75
VUS
20
Likely Benign
11
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
101
0
101
Likely Pathogenic
0
0
4
0
4
VUS
1
63
11
0
75
Likely Benign
0
6
2
12
20
Benign
0
8
1
2
11
Conflicting
2
Total17711914213

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

View in ClinVar →

Protein Context — Lollipop Plot

IRAK1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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