PLN

Chr 6AD

phospholamban

Also known as: CMD1P, CMH18, PLB

The protein encoded by this gene is found as a pentamer and is a major substrate for the cAMP-dependent protein kinase in cardiac muscle. The encoded protein is an inhibitor of cardiac muscle sarcoplasmic reticulum Ca(2+)-ATPase in the unphosphorylated state, but inhibition is relieved upon phosphorylation of the protein. The subsequent activation of the Ca(2+) pump leads to enhanced muscle relaxation rates, thereby contributing to the inotropic response elicited in heart by beta-agonists. The encoded protein is a key regulator of cardiac diastolic function. Mutations in this gene are a cause of inherited human dilated cardiomyopathy with refractory congestive heart failure, and also familial hypertrophic cardiomyopathy. [provided by RefSeq, Apr 2016]

GeneReviewsOMIMResearchGenerating clinical summary…
MultiplemechanismADLOEUF 1.562 OMIM phenotypes
Clinical SummaryPLN
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Gene-Disease Validity (ClinGen)
arrhythmogenic right ventricular cardiomyopathy · ADModerate

Moderate evidence — consider for supplementary testing

2 total gene-disease associations curated

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.00) despite low pLI — interpret in context.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — PLN
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
1.56LOEUF
pLI 0.449
Z-score 1.13
OE 0.00 (0.001.56)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
0.62Z-score
OE missense 0.67 (0.470.98)
19 obs / 28.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.00 (0.001.56)
00.351.4
Missense OE?0.67 (0.470.98)
00.61.4
Synonymous OE?0.53
01.21.6
LoF obs/exp: 0 / 1.5Missense obs/exp: 19 / 28.4Syn Z: 1.14
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePLN-related intrinsic cardiomyopathyOTHERAD

This gene — mechanism propensity

DN
0.7036th %ile
GOF
0.82top 10%
LOF
0.2776th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports dominant-negative. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median · 1 literature citation
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.

Literature Evidence

DNWhen combined with wild-type PLN to simulate heterozygous conditions, the mutants had a dominant negative effect on SERCA function.1

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

References

  1. 1.PMID 22427649

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PLN · protein map & ClinVar variants

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