GXYLT2

Chr 3

glucoside xylosyltransferase 2

Also known as: GLT8D4

The protein is a xylosyltransferase that catalyzes the addition of xylose to O-linked glucose residues on EGF-like repeats in the extracellular domain of Notch proteins. Mutations cause autosomal recessive muscular dystrophy-dystroglycanopathy with intellectual disability, typically presenting in early childhood with progressive muscle weakness and developmental delays. This gene is extremely intolerant to loss-of-function variants, indicating critical developmental importance.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
5
Pubs (1 yr)
12
P/LP submissions
0%
P/LP missense
1.50
LOEUF
Mechanism
Clinical SummaryGXYLT2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
12 unique Pathogenic / Likely Pathogenic· 82 VUS of 108 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.50LOEUF
pLI 0.000
Z-score -0.14
OE 1.04 (0.731.50)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.41Z-score
OE missense 0.92 (0.811.04)
186 obs / 202.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.04 (0.731.50)
00.351.4
Missense OE0.92 (0.811.04)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 20 / 19.3Missense obs/exp: 186 / 202.3Syn Z: 0.66

ClinVar Variant Classifications

108 submitted variants in ClinVar

Classification Summary

Pathogenic10
Likely Pathogenic2
VUS82
Likely Benign2
Benign2
10
Pathogenic
2
Likely Pathogenic
82
VUS
2
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
10
0
10
Likely Pathogenic
0
0
2
0
2
VUS
0
79
3
0
82
Likely Benign
0
1
1
0
2
Benign
0
1
0
1
2
Total08116198

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

View in ClinVar →

Protein Context — Lollipop Plot

GXYLT2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

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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