PIK3R3

Chr 1

phosphoinositide-3-kinase regulatory subunit 3

Also known as: p55, p55-GAMMA, p55PIK

The protein is a regulatory subunit of phosphatidylinositol 3-kinase (PI3K) that binds to activated protein-tyrosine kinases through its SH2 domains and regulates their activity in growth signaling pathways, including insulin signaling where it binds to IRS-1. Mutations cause autosomal recessive SHORT syndrome, characterized by short stature, hyperextensibility, ocular depression, Rieger anomaly, and teething delay. This gene is extremely intolerant to loss-of-function variants, indicating it is under strong evolutionary constraint.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.00
Clinical SummaryPIK3R3
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
1.00LOEUF
pLI 0.000
Z-score 1.54
OE 0.68 (0.481.00)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.04Z-score
OE missense 0.99 (0.891.10)
248 obs / 250.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.68 (0.481.00)
00.351.4
Missense OE0.99 (0.891.10)
00.61.4
Synonymous OE0.78
01.21.6
LoF obs/exp: 19 / 27.8Missense obs/exp: 248 / 250.0Syn Z: 1.59
DN
0.7228th %ile
GOF
0.6638th %ile
LOF
0.2969th %ile

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
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.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PIK3R3 · 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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