RPS6KA2

Chr 6

ribosomal protein S6 kinase A2

Also known as: HU-2, MAPKAPK1C, RSK, RSK3, S6K-alpha, S6K-alpha2, p90-RSK3, p90RSK2

The protein is a serine/threonine kinase that acts downstream of ERK signaling to regulate transcription factors, translation, and cellular proliferation, survival, and differentiation. Mutations cause Coffin-Lowry syndrome, an X-linked intellectual disability disorder characterized by distinctive facial features, skeletal abnormalities, and progressive intellectual disability that primarily affects males. The gene shows moderate constraint against loss-of-function variants, suggesting some intolerance to complete protein loss.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.45
Clinical SummaryRPS6KA2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.28) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.45LOEUF
pLI 0.009
Z-score 4.55
OE 0.28 (0.180.45)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.58Z-score
OE missense 0.66 (0.600.73)
310 obs / 467.0 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.28 (0.180.45)
00.351.4
Missense OE0.66 (0.600.73)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 13 / 46.4Missense obs/exp: 310 / 467.0Syn Z: 0.17
DN
0.76top 25%
GOF
0.6444th %ile
LOF
0.3356th %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

RPS6KA2 · 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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC