ICOS

Chr 2AR

inducible T cell costimulator

Also known as: AILIM, CD278, CVID1

The ICOS protein is a costimulatory receptor on activated T-cells that enhances T-cell responses to antigens, promotes T-cell and B-cell interactions, and is essential for normal antibody production and immune responses. Mutations cause common variable immunodeficiency, resulting in recurrent infections, hypogammaglobulinemia, and impaired antibody responses, with autosomal recessive inheritance. The gene shows low constraint to loss-of-function variants, which is consistent with the recessive inheritance pattern observed in affected patients.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismARLOEUF 0.911 OMIM phenotype
Clinical SummaryICOS
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Gene-Disease Validity (ClinGen)
common variable immunodeficiency · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.03) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
18 unique Pathogenic / Likely Pathogenic· 32 VUS of 100 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.91LOEUF
pLI 0.031
Z-score 1.78
OE 0.40 (0.190.91)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.21Z-score
OE missense 0.67 (0.550.81)
69 obs / 103.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.40 (0.190.91)
00.351.4
Missense OE0.67 (0.550.81)
00.61.4
Synonymous OE1.12
01.21.6
LoF obs/exp: 4 / 10.1Missense obs/exp: 69 / 103.6Syn Z: -0.55
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
moderateICOS-related combined immunodeficiency with recurrent sinopulmonary infections and recalcitrant wartsOTHERAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.76top 25%
GOF
0.6541th %ile
LOF
0.2288th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

ClinVar Variant Classifications

100 submitted variants in ClinVar

Classification Summary

Pathogenic17
Likely Pathogenic1
VUS32
Likely Benign44
Benign2
17
Pathogenic
1
Likely Pathogenic
32
VUS
44
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
6
0
11
0
17
Likely Pathogenic
1
0
0
0
1
VUS
0
26
6
0
32
Likely Benign
0
2
24
18
44
Benign
0
0
2
0
2
Total728431896

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

View in ClinVar →

Protein Context — Lollipop Plot

ICOS · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗