PLEC

Chr 8ARAD

plectin

Also known as: EBS1, EBS5A, EBS5B, EBS5C, EBS5D, EBSMD, EBSND, EBSO

Plectin interlinks intermediate filaments with microtubules and microfilaments, anchors intermediate filaments to desmosomes and hemidesmosomes, and serves as a structural component of muscle. Mutations cause epidermolysis bullosa simplex with various presentations (including muscular dystrophy, pyloric atresia, and Ogna type) and limb-girdle muscular dystrophy, inherited in autosomal recessive or autosomal dominant patterns. The gene is highly constrained against loss-of-function mutations, reflecting its essential role in maintaining skin and muscle tissue integrity.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismAR/ADLOEUF 0.395 OMIM phenotypes
Clinical SummaryPLEC
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Gene-Disease Validity (ClinGen)
PLEC-related muscular dystrophy-epidermolysis bullosa simplex spectrum disorder · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.31) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.39LOEUF
pLI 0.000
Z-score 8.45
OE 0.31 (0.250.39)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-2.57Z-score
OE missense 1.13 (1.101.16)
3516 obs / 3113.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.31 (0.250.39)
00.351.4
Missense OE1.13 (1.101.16)
00.61.4
Synonymous OE1.38
01.21.6
LoF obs/exp: 55 / 175.9Missense obs/exp: 3516 / 3113.0Syn Z: -11.32
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongPLEC-related epidermolysis bullosa simplex and limb-girdle muscular dystrophyLOFAR
strongPLEC-related epidermolysis bullosa simplex (Ogna)OTHERAD
DN
0.79top 25%
GOF
0.79top 25%
LOF
0.2190th %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 · 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

DNAutosomal dominant EBS is associated with either a heterozygous dominant-negative variant in KRT5, KRT14, or PLEC or a heterozygous pathogenic variant in KLHL24.PMID:20301543

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PLEC · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗