IRS1

Chr 2AD

insulin receptor substrate 1

Also known as: HIRS-1

The insulin receptor substrate 1 protein serves as a signaling adapter that is phosphorylated by insulin and insulin-like growth factor I receptors, functioning as a scaffold to propagate downstream signaling pathways critical for glucose homeostasis, lipid metabolism, growth, and development. Mutations cause autosomal dominant susceptibility to type 2 diabetes mellitus and insulin resistance. This gene is not highly constrained against loss-of-function variants, consistent with its role in disease susceptibility rather than severe developmental disorders.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

{Type 2 diabetes mellitus, susceptibility to}MIM #125853
AD
1
Active trials
193
Pubs (1 yr)
26
P/LP submissions
4%
P/LP missense
0.50
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryIRS1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.29) despite low pLI — interpret in context.
📋
ClinVar Variants
26 unique Pathogenic / Likely Pathogenic· 168 VUS of 232 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
📖
GeneReview available — IRS1
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.50LOEUF
pLI 0.024
Z-score 3.69
OE 0.29 (0.170.50)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.07Z-score
OE missense 0.99 (0.941.05)
787 obs / 792.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.29 (0.170.50)
00.351.4
Missense OE0.99 (0.941.05)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 9 / 31.3Missense obs/exp: 787 / 792.4Syn Z: 0.12
DN
0.6260th %ile
GOF
0.4874th %ile
LOF
0.51top 25%

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

DNprediction above median
GOF1 literature citation

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.

Literature Evidence

GOFGenomic analysis of de novo vemurafenib-resistant cHCL identified a novel gain-of-function mutation in IRS1 and losses of NF1 and NF2, each of which contributed to resistance.PMID:28801450

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

232 submitted variants in ClinVar

Classification Summary

Pathogenic24
Likely Pathogenic2
VUS168
Likely Benign18
Benign13
Conflicting1
24
Pathogenic
2
Likely Pathogenic
168
VUS
18
Likely Benign
13
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
1
23
0
24
Likely Pathogenic
0
0
2
0
2
VUS
0
164
4
0
168
Likely Benign
0
7
2
9
18
Benign
0
4
1
8
13
Conflicting
1
Total01763217226

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

View in ClinVar →

Protein Context — Lollipop Plot

IRS1 · protein map & ClinVar variants

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

Clinical Literature
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