TJP1

Chr 15

tight junction protein 1

Also known as: ZO-1

TJP1 encodes a tight junction scaffolding protein that links transmembrane proteins to the actin cytoskeleton, regulating the movement of substances between epithelial and endothelial cells and maintaining cell polarity. Mutations cause autosomal dominant progressive familial intrahepatic cholestasis type 4, which presents in infancy with cholestasis, pruritus, and progressive liver disease. This gene is extremely intolerant to loss-of-function variants (pLI ~1.0, LOEUF 0.071), indicating that haploinsufficiency is likely pathogenic.

OMIMResearchSummary from RefSeq, UniProt
LOFmechanismLOEUF 0.07
Clinical SummaryTJP1
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Gene-Disease Validity (ClinGen)
arrhythmogenic right ventricular cardiomyopathy · ADLimited

Limited evidence — not for standalone diagnostic reporting

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.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.07LOEUF
pLI 1.000
Z-score 8.52
OE 0.02 (0.010.07)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
1.54Z-score
OE missense 0.86 (0.810.91)
829 obs / 963.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.02 (0.010.07)
00.351.4
Missense OE0.86 (0.810.91)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 2 / 88.4Missense obs/exp: 829 / 963.7Syn Z: -0.30
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedTJP1-related arrhythmogenic right ventricular cardiomyopathyOTHERAD
DN
0.3495th %ile
GOF
0.4382th %ile
LOF
0.80top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.07

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

TJP1 · 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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