ATP6V0A4

Chr 7AR

ATPase H+ transporting V0 subunit a4

The protein is a subunit of the vacuolar H+-ATPase complex that acidifies intracellular compartments and mediates vectorial acid transport into urine by the kidney. Autosomal recessive mutations cause distal renal tubular acidosis type 3, with or without sensorineural hearing loss, through a predicted gain-of-function mechanism.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismARLOEUF 1.001 OMIM phenotype
Clinical SummaryATP6V0A4
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.00LOEUF
pLI 0.000
Z-score 1.53
OE 0.76 (0.591.00)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.85Z-score
OE missense 0.89 (0.820.96)
408 obs / 459.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.76 (0.591.00)
00.351.4
Missense OE0.89 (0.820.96)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 37 / 48.5Missense obs/exp: 408 / 459.6Syn Z: 0.08
DN
0.6839th %ile
GOF
0.7127th %ile
LOF
0.2873th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
DNprediction 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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ATP6V0A4 · 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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