TM4SF5

Chr 17

transmembrane 4 L six family member 5

TM4SF5 encodes a transmembrane glycoprotein that acts as a lysosomal membrane arginine sensor and forms complexes with mTOR and SLC38A9 to regulate mTORC1 activation and cell cycle progression. The gene shows low constraint against loss-of-function variants (pLI 0.008, LOEUF 0.97), and no definitive Mendelian diseases have been established from TM4SF5 mutations in current medical literature. Overexpression has been associated with tumor cell growth, but pathogenic germline variants causing pediatric neurological conditions have not been well-characterized.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.97
Clinical SummaryTM4SF5
Population Constraint (gnomAD)
Low constraint (pLI 0.01) — 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
0.97LOEUF
pLI 0.008
Z-score 1.64
OE 0.46 (0.240.97)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.79Z-score
OE missense 0.81 (0.690.95)
108 obs / 133.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.46 (0.240.97)
00.351.4
Missense OE0.81 (0.690.95)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 5 / 10.8Missense obs/exp: 108 / 133.9Syn Z: 0.52
DN
0.7133th %ile
GOF
0.80top 10%
LOF
0.2583th %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

TM4SF5 · 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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