CLDN4

Chr 7

claudin 4

Also known as: CPE-R, CPER, CPETR, CPETR1, WBSCR8, hCPE-R

Claudin-4 is an integral membrane protein that regulates epithelial tight junction barrier properties and paracellular ion transport, particularly chloride permeability in renal collecting ducts. CLDN4 is deleted in Williams-Beuren syndrome, a multisystem neurodevelopmental disorder. The gene shows low constraint to loss-of-function variation (LOEUF 1.827), suggesting that complete loss may be tolerated when occurring as part of larger deletions.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.83
Clinical SummaryCLDN4
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.83LOEUF
pLI 0.000
Z-score -0.17
OE 1.08 (0.581.83)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.76Z-score
OE missense 0.83 (0.710.96)
124 obs / 150.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.08 (0.581.83)
00.351.4
Missense OE0.83 (0.710.96)
00.61.4
Synonymous OE1.14
01.21.6
LoF obs/exp: 6 / 5.6Missense obs/exp: 124 / 150.2Syn Z: -0.93
DN
0.78top 25%
GOF
0.79top 25%
LOF
0.2582th %ile

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

GOFprediction above median
DNprediction 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

CLDN4 · 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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