GRHL2

Chr 8ADAR

grainyhead like transcription factor 2

Also known as: BOM, DFNA28, ECTDS, PPCD4, TFCP2L3

This gene encodes a transcription factor that binds to specific DNA sequences to regulate epithelial development, primary neurulation, and expression of tight junction proteins including claudins. Mutations cause autosomal dominant non-syndromic sensorineural deafness (DFNA28), posterior polymorphous corneal dystrophy type 4, and autosomal recessive ectodermal dysplasia with short stature. The pathogenic mechanism involves loss of function, as the protein is highly intolerant to loss-of-function variants.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismAD/ARLOEUF 0.273 OMIM phenotypes
Clinical SummaryGRHL2
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Gene-Disease Validity (ClinGen)
nonsyndromic genetic hearing loss · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
21 unique Pathogenic / Likely Pathogenic· 112 VUS of 200 total submissions
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GeneReview available — GRHL2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.27LOEUF
pLI 0.997
Z-score 5.04
OE 0.13 (0.070.27)
Highly constrained

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

Missense Constraint
1.93Z-score
OE missense 0.71 (0.640.79)
246 obs / 347.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.13 (0.070.27)
00.351.4
Missense OE0.71 (0.640.79)
00.61.4
Synonymous OE1.14
01.21.6
LoF obs/exp: 5 / 39.0Missense obs/exp: 246 / 347.0Syn Z: -1.27
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongGRHL2-related posterior polymorphous corneal dystrophyOTHERAD
strongGRHL2-related ectodermal dysplasia and short stature syndromeOTHERAR
DN
0.3793th %ile
GOF
0.2895th %ile
LOF
0.78top 5%

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

LOFprediction above median · 1 literature citation · 81% of P/LP variants are LoF · LOEUF 0.27

Literature Evidence

LOFHaploinsufficiency of the GRHL2 gene contained within the microdeletion is proposed as a candidate genetic mechanism for this patient's hearing loss.PMID:26455667

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

200 submitted variants in ClinVar

Classification Summary

Pathogenic13
Likely Pathogenic8
VUS112
Likely Benign50
Benign1
13
Pathogenic
8
Likely Pathogenic
112
VUS
50
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
9
0
4
0
13
Likely Pathogenic
8
0
0
0
8
VUS
1
101
9
1
112
Likely Benign
0
2
27
21
50
Benign
0
0
0
1
1
Total181034023184

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

View in ClinVar →

Protein Context — Lollipop Plot

GRHL2 · 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 →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗