GYG2

Chr X

glycogenin 2

Also known as: GN-2, GN2

This X-linked gene encodes glycogenin, which catalyzes the formation of short glucose chains that serve as primers for glycogen synthesis, primarily in liver and playing a role in blood glucose homeostasis. Mutations cause glycogen storage disease type XV, characterized by hepatomegaly, fasting hypoglycemia, and elevated liver enzymes typically presenting in infancy or early childhood. The gene shows very low constraint against loss-of-function variants, and inheritance follows an X-linked pattern.

OMIMResearchSummary from RefSeq, UniProt
GOFmechanismLOEUF 1.57
Clinical SummaryGYG2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
95 unique Pathogenic / Likely Pathogenic· 117 VUS of 400 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.57LOEUF
pLI 0.000
Z-score -0.03
OE 1.01 (0.671.57)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.08Z-score
OE missense 0.98 (0.881.11)
199 obs / 202.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.01 (0.671.57)
00.351.4
Missense OE0.98 (0.881.11)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 14 / 13.9Missense obs/exp: 199 / 202.0Syn Z: -0.02
DN
0.5968th %ile
GOF
0.6639th %ile
LOF
0.3453th %ile

The highest-scoring mechanism for this gene is gain-of-function.

GOFprediction 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

400 submitted variants in ClinVar

Classification Summary

Pathogenic92
Likely Pathogenic3
VUS117
Likely Benign84
Benign54
Conflicting10
92
Pathogenic
3
Likely Pathogenic
117
VUS
84
Likely Benign
54
Benign
10
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
92
0
92
Likely Pathogenic
0
0
3
0
3
VUS
5
76
35
1
117
Likely Benign
0
6
50
28
84
Benign
0
6
43
5
54
Conflicting
10
Total58822334360

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

View in ClinVar →

Protein Context — Lollipop Plot

GYG2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC