IRS4

Chr XXLR

insulin receptor substrate 4

Also known as: CHNG9, IRS-4, PY160

IRS4 encodes insulin receptor substrate 4, a cytoplasmic signaling protein that serves as an interface between growth factor receptors (including insulin, IGF1, and FGFR1 receptors) and downstream SH2 domain-containing signaling molecules involved in growth, reproduction, and glucose homeostasis. Mutations cause congenital nongoitrous hypothyroidism type 9 with X-linked recessive inheritance. The gene shows significant constraint against loss-of-function variants (LOEUF 0.43), suggesting intolerance to protein-disrupting mutations.

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

Hypothyroidism, congenital, nongoitrous, 9MIM #301035
XLR
0
Active trials
9
Pubs (1 yr)
87
P/LP submissions
0%
P/LP missense
LOEUF
LOF
Mechanism· predicted
Clinical SummaryIRS4
📋
ClinVar Variants
82 unique Pathogenic / Likely Pathogenic· 136 VUS of 243 total submissions
Some data sources returned errors (1)

gnomad: TimeoutError: The operation was aborted due to timeout

Population Genetics & Constraint

Constraint data not available from gnomAD.

DN
0.4388th %ile
GOF
0.3986th %ile
LOF
0.67top 25%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median

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

ClinVar Variant Classifications

243 submitted variants in ClinVar

Classification Summary

Pathogenic79
Likely Pathogenic3
VUS136
Likely Benign19
Benign3
Conflicting2
79
Pathogenic
3
Likely Pathogenic
136
VUS
19
Likely Benign
3
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
79
0
79
Likely Pathogenic
0
0
3
0
3
VUS
1
131
4
0
136
Likely Benign
0
13
0
6
19
Benign
0
2
0
1
3
Conflicting
2
Total1146867242

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

View in ClinVar →

Protein Context — Lollipop Plot

IRS4 · 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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