TMEM88 encodes a protein that inhibits the Wnt/beta-catenin signaling pathway and is crucial for heart development, acting downstream of GATA factors in cardiac lineage specification. Mutations cause autosomal recessive congenital heart disease with onset in the neonatal period. The gene shows moderate tolerance to loss-of-function variants based on population genetics data.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.25
Clinical SummaryTMEM88
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.27) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.25LOEUF
pLI 0.277
Z-score 1.31
OE 0.27 (0.091.25)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.34Z-score
OE missense 0.91 (0.771.08)
94 obs / 103.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.27 (0.091.25)
00.351.4
Missense OE0.91 (0.771.08)
00.61.4
Synonymous OE1.13
01.21.6
LoF obs/exp: 1 / 3.7Missense obs/exp: 94 / 103.6Syn Z: -0.72
DN
0.6453th %ile
GOF
0.84top 5%
LOF
0.3066th %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

TMEM88 · 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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