YWHAG

Chr 7AD

tyrosine 3-monooxygenase/tryptophan 5-monooxygenase activation protein gamma

Also known as: 14-3-3GAMMA, DEE56, EIEE56, PPP1R170

The protein belongs to the 14-3-3 family and mediates signal transduction by binding to phosphoserine-containing proteins, with particular expression in muscle tissues and interactions with RAF1 and protein kinase C. Mutations cause developmental and epileptic encephalopathy 56 through an autosomal dominant inheritance pattern. The high pLI score (0.96) and low LOEUF score (0.29) indicate the gene is highly intolerant to loss-of-function variants, suggesting haploinsufficiency as the likely pathogenic mechanism.

OMIMResearchSummary from RefSeq, OMIM, UniProt
GOFmechanismADLOEUF 0.291 OMIM phenotype
Clinical SummaryYWHAG
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.96). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
58 unique Pathogenic / Likely Pathogenic· 103 VUS of 279 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.29LOEUF
pLI 0.964
Z-score 2.98
OE 0.00 (0.000.29)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.95Z-score
OE missense 0.34 (0.270.43)
54 obs / 158.6 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.00 (0.000.29)
00.351.4
Missense OE0.34 (0.270.43)
00.61.4
Synonymous OE1.26
01.21.6
LoF obs/exp: 0 / 10.3Missense obs/exp: 54 / 158.6Syn Z: -1.71
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongYWHAG-related early-onset epilepsyGOFAD
DN
0.5082th %ile
GOF
0.4381th %ile
LOF
0.58top 25%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOF1 literature citation · 28% of P/LP variants are LoF · LOEUF 0.29

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.

Literature Evidence

LOFTwo unrelated human patients with interstitial deletions at chromosome 7q11.23 (613729) including the YWHAG gene had severe infantile seizures and hypertrophic cardiomyopathy, which Komoike et al. (2010) postulated may have resulted from haploinsufficiency of YWHAG.PMID:20146355

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

279 submitted variants in ClinVar

Classification Summary

Pathogenic43
Likely Pathogenic15
VUS103
Likely Benign103
Benign8
Conflicting4
43
Pathogenic
15
Likely Pathogenic
103
VUS
103
Likely Benign
8
Benign
4
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
13
7
23
0
43
Likely Pathogenic
3
8
4
0
15
VUS
8
75
20
0
103
Likely Benign
0
2
10
91
103
Benign
0
1
2
5
8
Conflicting
4
Total24935996276

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

View in ClinVar →

Protein Context — Lollipop Plot

YWHAG · 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
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