GYG1

Chr 3AR

glycogenin 1

Also known as: GSD15, GYG

This gene encodes a member of the glycogenin family. Glycogenin is a glycosyltransferase that catalyzes the formation of a short glucose polymer from uridine diphosphate glucose in an autoglucosylation reaction. This reaction is followed by elongation and branching of the polymer, catalyzed by glycogen synthase and branching enzyme, to form glycogen. This gene is expressed in muscle and other tissues. Mutations in this gene result in glycogen storage disease XV. This gene has pseudogenes on chromosomes 1, 8 and 13 respectively. Alternatively spliced transcript variants encoding different isoforms have been identified.[provided by RefSeq, Sep 2010]

OMIMResearchGenerating clinical summary…
GOFmechanismARLOEUF 1.412 OMIM phenotypes
Clinical SummaryGYG1
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Gene-Disease Validity (ClinGen)
polyglucosan body myopathy type 2 · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
41 unique Pathogenic / Likely Pathogenic· 145 VUS of 367 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
1.41LOEUF
pLI 0.000
Z-score 0.21
OE 0.95 (0.651.41)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
0.06Z-score
OE missense 0.99 (0.881.11)
185 obs / 187.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.95 (0.651.41)
00.351.4
Missense OE?0.99 (0.881.11)
00.61.4
Synonymous OE?0.90
01.21.6
LoF obs/exp: 18 / 19.0Missense obs/exp: 185 / 187.5Syn Z: 0.70

This gene — mechanism propensity

DN
0.5771th %ile
GOF
0.6639th %ile
LOF
0.3745th %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

367 submitted variants in ClinVar

Classification Summary

Pathogenic24
Likely Pathogenic17
VUS145
Likely Benign132
Benign34
Conflicting5
24
Pathogenic
17
Likely Pathogenic
145
VUS
132
Likely Benign
34
Benign
5
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
22
0
2
0
24
Likely Pathogenic
14
2
1
0
17
VUS
4
136
5
0
145
Likely Benign
0
15
62
55
132
Benign
0
1
29
4
34
Conflicting
5
Total401549959357

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

View in ClinVar →

21 pathogenic / likely-pathogenic (of 24) ClinVar copy-number / structural variants overlap GYG1 — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

GYG1 · 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.

Search ClinicalTrials.gov →