ATP4B

Chr 13

ATPase H+/K+ transporting subunit beta

Also known as: ATP6B

The beta subunit of gastric H+/K+-ATPase functions as a proton pump that exchanges hydrogen and potassium ions across the membrane of gastric parietal cells, playing structural and regulatory roles in gastric acid secretion. Mutations cause congenital chloride diarrhea with gastric hypochlorhydria, inherited in an autosomal recessive pattern. This gene shows low constraint against loss-of-function variants (pLI near 0), consistent with recessive inheritance where heterozygous carriers are typically unaffected.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
6
Pubs (1 yr)
116
P/LP submissions
0%
P/LP missense
1.05
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryATP4B
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
116 unique Pathogenic / Likely Pathogenic· 64 VUS of 191 total submissions
Some data sources returned errors (1)

ensembl: TimeoutError: The operation was aborted due to timeout

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.05LOEUF
pLI 0.000
Z-score 1.42
OE 0.60 (0.361.05)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.17Z-score
OE missense 0.96 (0.851.10)
169 obs / 175.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.60 (0.361.05)
00.351.4
Missense OE0.96 (0.851.10)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 9 / 14.9Missense obs/exp: 169 / 175.2Syn Z: -0.05
DN
0.7230th %ile
GOF
0.6735th %ile
LOF
0.1895th %ile

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

DNprediction above median
GOFprediction 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

191 submitted variants in ClinVar

Classification Summary

Pathogenic113
Likely Pathogenic3
VUS64
Likely Benign6
113
Pathogenic
3
Likely Pathogenic
64
VUS
6
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
113
0
113
Likely Pathogenic
0
0
3
0
3
VUS
0
50
14
0
64
Likely Benign
0
6
0
0
6
Benign
0
0
0
0
0
Total0561300186

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

View in ClinVar →

Protein Context — Lollipop Plot

ATP4B · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

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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