TJP2

Chr 9AR

tight junction protein 2

Also known as: C9DUPq21.11, DFNA51, DUP9q21.11, FHCA1, PFIC4, X104, ZO2

The protein functions as a component of tight junction barriers in epithelial and endothelial cells and is necessary for proper tight junction assembly. Mutations cause progressive familial intrahepatic cholestasis type 4 and familial hypercholanemia, both involving hepatic bile acid metabolism abnormalities. This gene follows autosomal recessive inheritance and is highly constrained against loss-of-function variation (pLI near 0, LOEUF 0.635).

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismARLOEUF 0.642 OMIM phenotypes
Clinical SummaryTJP2
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Gene-Disease Validity (ClinGen)
nonsyndromic genetic hearing loss · ADLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.64LOEUF
pLI 0.000
Z-score 3.87
OE 0.46 (0.340.64)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.57Z-score
OE missense 0.94 (0.881.00)
700 obs / 743.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.46 (0.340.64)
00.351.4
Missense OE0.94 (0.881.00)
00.61.4
Synonymous OE0.94
01.21.6
LoF obs/exp: 28 / 60.4Missense obs/exp: 700 / 743.5Syn Z: 0.76
DN
0.6552th %ile
GOF
0.6248th %ile
LOF
0.4430th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

TJP2 · 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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