TMTC3

Chr 12AR

transmembrane O-mannosyltransferase targeting cadherins 3

Also known as: LIS8, SMILE

This protein functions as an O-mannosyl-transferase that transfers mannose residues to serine or threonine residues, primarily on cadherin superfamily proteins, and regulates proteasomal protein degradation in the endoplasmic reticulum. Mutations cause autosomal recessive lissencephaly 8, a severe brain malformation disorder affecting cortical development. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.56), consistent with its role in essential cellular glycosylation processes.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.561 OMIM phenotype
Clinical SummaryTMTC3
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

Moderate LoF intolerance
LoF Constraint
0.56LOEUF
pLI 0.000
Z-score 3.76
OE 0.37 (0.240.56)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.62Z-score
OE missense 0.79 (0.720.86)
364 obs / 461.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.37 (0.240.56)
00.351.4
Missense OE0.79 (0.720.86)
00.61.4
Synonymous OE0.89
01.21.6
LoF obs/exp: 15 / 41.0Missense obs/exp: 364 / 461.7Syn Z: 1.11
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongTMTC3-related cobblestone lissencephalyLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.76top 25%
GOF
0.7029th %ile
LOF
0.2679th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

TMTC3 · 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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