RYR2

Chr 1AD

ryanodine receptor 2

Also known as: ARVC2, ARVD2, RYR-2, RyR, VACRDS, VTSIP

This gene encodes a cardiac ryanodine receptor that functions as a cytosolic calcium-activated calcium channel, mediating calcium release from the sarcoplasmic reticulum to trigger cardiac muscle contraction. Mutations cause catecholaminergic polymorphic ventricular tachycardia and arrhythmogenic right ventricular dysplasia with autosomal dominant inheritance. The gene is highly constrained against loss-of-function variants, indicating that haploinsufficiency is likely not tolerated.

OMIMResearchSummary from RefSeq, OMIM, UniProt
GOFmechanismADLOEUF 0.252 OMIM phenotypes
Clinical SummaryRYR2
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Gene-Disease Validity (ClinGen)
hypertrophic cardiomyopathy · ADLimited

Limited evidence — not for standalone diagnostic reporting

4 total gene-disease associations curated

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.25LOEUF
pLI 1.000
Z-score 11.84
OE 0.20 (0.160.25)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
5.78Z-score
OE missense 0.68 (0.660.71)
1804 obs / 2639.8 exp
Constrained

Extremely missense-constrained (top ~0.01%)

Observed / Expected Ratios
LoF OE0.20 (0.160.25)
00.351.4
Missense OE0.68 (0.660.71)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 50 / 253.4Missense obs/exp: 1804 / 2639.8Syn Z: -0.20
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveRYR2-related catecholaminergic polymorphic ventricular tachycardiaGOFAD
limitedRYR2-related hypertrophic cardiomyopathyOTHERAD
limitedRYR2-related arrhythmogenic right ventricular cardiomyopathyOTHERAD
limitedRYR2-related catecholaminergic polymorphic ventricular tachycardia and intellectual disabilityOTHERAD
DN
0.4586th %ile
GOF
0.5563th %ile
LOF
0.63top 25%

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

LOFLOEUF 0.25
GOF1 literature citation
DN1 literature citation

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.

Literature Evidence

DNThe G4935R mutation completely abolished caffeine activation of and [3H]ryanodine binding to RyR2. Co-expression studies showed that the G4935R mutation exerted dominant negative impact on the RyR2 wildtype (WT) channel.PMID:33825858
GOFUnlike the gain-of-function mechanism observed in RYR2-mediated CPVT, the homozygous multiexon duplication precipitated a dramatic reduction in RYR2 transcription and RyR2 protein translation, a loss of function in calcium handling, and a calcium-induced calcium release apparatus that is insensitivePMID:32663189

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

RYR2 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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