LAGE3

Chr XXLR

L antigen family member 3

Also known as: CVG5, DXS9879E, DXS9951E, ESO3, GAMOS2, ITBA2, Pcc1

LAGE3 encodes a component of the EKC/KEOPS complex that functions as a dimerization module and is required for threonylcarbamoyl modification of specific tRNAs. Mutations cause Galloway-Mowat syndrome 2, a disorder affecting the kidneys and nervous system with X-linked recessive inheritance. The gene shows relatively low constraint scores, consistent with recessive disease mechanisms.

OMIMResearchSummary from RefSeq, OMIM, UniProt
XLRLOEUF 1.811 OMIM phenotype
Clinical SummaryLAGE3
Population Constraint (gnomAD)
Low constraint (pLI 0.13) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
83 unique Pathogenic / Likely Pathogenic· 66 VUS of 200 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.81LOEUF
pLI 0.127
Z-score 0.49
OE 0.60 (0.191.81)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.41Z-score
OE missense 0.84 (0.671.08)
46 obs / 54.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.60 (0.191.81)
00.351.4
Missense OE0.84 (0.671.08)
00.61.4
Synonymous OE0.85
01.21.6
LoF obs/exp: 1 / 1.7Missense obs/exp: 46 / 54.5Syn Z: 0.57

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic80
Likely Pathogenic3
VUS66
Likely Benign19
Benign10
Conflicting5
80
Pathogenic
3
Likely Pathogenic
66
VUS
19
Likely Benign
10
Benign
5
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
2
77
0
80
Likely Pathogenic
0
0
3
0
3
VUS
4
48
14
0
66
Likely Benign
0
2
1
16
19
Benign
0
5
4
1
10
Conflicting
5
Total5579917183

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

LAGE3 · 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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