CLDN5

Chr 22

claudin 5

Also known as: AWAL, BEC1, CPETRL1, TMDVCF, TMVCF

This gene encodes claudin-5, an integral membrane protein that forms tight junction strands and plays a major role in obliterating the intercellular space to prevent solutes and water from passing freely between endothelial cell sheets. Mutations cause velocardiofacial syndrome, which follows an autosomal dominant inheritance pattern. The gene is highly constrained against loss-of-function variants (pLI 0.90, LOEUF 0.41), indicating that complete loss of protein function is likely not tolerated.

OMIMResearchSummary from RefSeq, UniProt
GOFmechanismLOEUF 0.41
Clinical SummaryCLDN5
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.90) — some intolerance to loss-of-function variants.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.41LOEUF
pLI 0.895
Z-score 2.50
OE 0.00 (0.000.41)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.44Z-score
OE missense 0.68 (0.580.80)
108 obs / 159.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.41)
00.351.4
Missense OE0.68 (0.580.80)
00.61.4
Synonymous OE0.94
01.21.6
LoF obs/exp: 0 / 7.3Missense obs/exp: 108 / 159.0Syn Z: 0.38
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedCLDN5-related neurodevelopmental disorderOTHERAD
DN
0.5772th %ile
GOF
0.73top 25%
LOF
0.4528th %ile

The highest-scoring mechanism for this gene is gain-of-function.

GOFprediction above median · 1 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

GOFThis mutation is the first gain-of-function mutation identified in the CLDN gene family with all others representing loss-of-function mutations resulting in mis-localization of CLDN protein and/or attenuated barrier function.PMID:36978081

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CLDN5 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
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