CHAC2

Chr 2

ChaC glutathione specific gamma-glutamylcyclotransferase 2

Also known as: GCG1

The gamma-glutamyl cyclotransferase CHAC2 catalyzes the cleavage of glutathione into 5-oxo-L-proline and cysteinyl-glycine, and is upregulated during endoplasmic reticulum stress to promote apoptosis through glutathione depletion. Mutations cause autosomal recessive early infantile epileptic encephalopathy, characterized by severe seizures and developmental delays beginning in infancy. This gene is highly intolerant to loss-of-function variants, suggesting that complete loss of function is likely pathogenic.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
5
Pubs (1 yr)
10
P/LP submissions
0%
P/LP missense
1.84
LOEUF
Mechanism
Clinical SummaryCHAC2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 49 VUS of 66 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.84LOEUF
pLI 0.000
Z-score -0.38
OE 1.16 (0.671.84)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-1.18Z-score
OE missense 1.34 (1.161.54)
131 obs / 98.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.16 (0.671.84)
00.351.4
Missense OE1.34 (1.161.54)
00.61.4
Synonymous OE1.28
01.21.6
LoF obs/exp: 8 / 6.9Missense obs/exp: 131 / 98.0Syn Z: -1.28

ClinVar Variant Classifications

66 submitted variants in ClinVar

Classification Summary

Pathogenic9
Likely Pathogenic1
VUS49
Benign1
9
Pathogenic
1
Likely Pathogenic
49
VUS
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
9
0
9
Likely Pathogenic
0
0
1
0
1
VUS
0
47
2
0
49
Likely Benign
0
0
0
0
0
Benign
0
0
1
0
1
Total04713060

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

View in ClinVar →

Protein Context — Lollipop Plot

CHAC2 · 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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