ADGRV1

Chr 5ADARDigenic dominant

adhesion G protein-coupled receptor V1

The protein is a G-protein coupled receptor essential for hearing and vision development that regulates cAMP signaling, hair bundle formation in cochlear cells, and photoreceptor function. Mutations cause Usher syndrome type 2C (progressive hearing loss and retinitis pigmentosa) and familial febrile seizures, with inheritance patterns including autosomal recessive, autosomal dominant, and digenic forms. The gene is not highly constrained against loss-of-function variants and affects multiple systems including auditory, visual, and neurological function.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismAD/AR/Digenic dominantLOEUF 0.523 OMIM phenotypes
Clinical SummaryADGRV1
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Gene-Disease Validity (ClinGen)
nonsyndromic genetic hearing loss · ARDisputed

Disputed — evidence questions this relationship

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
Some data sources returned errors (1)

ncbi: Error: NCBI fetch failed: 429 https://eutils.ncbi.nlm.nih.gov/entrez/eutils/esearch.fcgi

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.52LOEUF
pLI 0.000
Z-score 8.19
OE 0.44 (0.380.52)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.07Z-score
OE missense 1.00 (0.971.03)
3130 obs / 3141.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.44 (0.380.52)
00.351.4
Missense OE1.00 (0.971.03)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 110 / 249.6Missense obs/exp: 3130 / 3141.7Syn Z: -0.55
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongADGRV1-related Usher syndromeLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6936th %ile
GOF
0.6051th %ile
LOF
0.3453th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Literature Evidence

LOFIn patients with 5q14.3 deletion and epilepsy, ADGRV1 haploinsufficiency likely contributes to seizure development.PMID:29266188

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ADGRV1 · 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.

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Clinical Literature
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