ATP6V1B1

Chr 2AR

ATPase H+ transporting V1 subunit B1

Also known as: ATP6B1, DRTA2, RTA1B, VATB, VMA2, VPP3

This gene encodes the B1 subunit of vacuolar H+-ATPase, a multisubunit enzyme that acidifies intracellular compartments and, in renal intercalated cells, secretes protons into urine for urinary acidification. Mutations cause autosomal recessive distal renal tubular acidosis with sensorineural hearing loss, a condition affecting kidney function and hearing. The gene shows minimal constraint against loss-of-function variants (pLI near zero), consistent with its recessive inheritance pattern.

OMIMResearchSummary from RefSeq, UniProt
LOFmechanismARLOEUF 0.951 OMIM phenotype
Clinical SummaryATP6V1B1
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Gene-Disease Validity (ClinGen)
renal tubular acidosis, distal, 2, with progressive sensorineural hearing loss · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

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
0.95LOEUF
pLI 0.000
Z-score 1.77
OE 0.64 (0.440.95)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.23Z-score
OE missense 0.96 (0.881.06)
297 obs / 308.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.64 (0.440.95)
00.351.4
Missense OE0.96 (0.881.06)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 18 / 28.2Missense obs/exp: 297 / 308.5Syn Z: 0.37
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveATP6V1B1-related distal renal tubular acidosis with deafnessLOFAR

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

DN
0.78top 25%
GOF
0.5954th %ile
LOF
0.3068th %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.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ATP6V1B1 · 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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