ICOSLG

Chr 21AR

inducible T cell costimulator ligand

Also known as: B7-H2, B7H2, B7RP-1, B7RP1, B7h, CD275, GL50, ICOS-L

ICOSLG encodes a ligand for the T-cell surface receptor ICOS that provides costimulatory signals for T-cell proliferation and cytokine secretion, induces B-cell proliferation and differentiation into plasma cells, and is required for proper neutrophil transmigration in endothelial cells. Mutations cause autosomal recessive immunodeficiency 119, affecting the immune system. The gene shows low constraint to loss-of-function variants (pLI 0.0004, LOEUF 0.92), consistent with recessive inheritance where heterozygous carriers are typically unaffected.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

?Immunodeficiency 119MIM #620825
AR
0
Active trials
12
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.92
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryICOSLG
🧬
Gene-Disease Validity (ClinGen)
combined immunodeficiency · ARModerate

Moderate evidence — consider for supplementary testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.92LOEUF
pLI 0.000
Z-score 1.80
OE 0.51 (0.300.92)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.13Z-score
OE missense 0.77 (0.680.89)
153 obs / 197.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.51 (0.300.92)
00.351.4
Missense OE0.77 (0.680.89)
00.61.4
Synonymous OE1.11
01.21.6
LoF obs/exp: 8 / 15.7Missense obs/exp: 153 / 197.7Syn Z: -0.82
DN
0.6938th %ile
GOF
0.81top 10%
LOF
0.2091th %ile

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

GOFprediction above median
DNprediction 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.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ICOSLG · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC