ATP2B3

Chr XXLR

ATPase plasma membrane Ca2+ transporting 3

Also known as: CFAP39, CLA2, OPCA, PMCA3, PMCA3a, SCAX1

This gene encodes plasma membrane calcium ATPase isoform 3, which uses ATP to transport cytosolic calcium ions across the plasma membrane to maintain basal intracellular calcium levels at presynaptic terminals. Mutations cause X-linked spinocerebellar ataxia type 1 through an X-linked recessive inheritance pattern. The pathogenic mechanism appears to involve gain-of-function effects that disrupt normal calcium homeostasis at synapses.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismXLRLOEUF 0.221 OMIM phenotype
Clinical SummaryATP2B3
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
67 unique Pathogenic / Likely Pathogenic· 130 VUS of 300 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.22LOEUF
pLI 0.999
Z-score 5.00
OE 0.09 (0.040.22)
Highly constrained

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

Missense Constraint
2.75Z-score
OE missense 0.68 (0.630.74)
405 obs / 593.2 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.09 (0.040.22)
00.351.4
Missense OE0.68 (0.630.74)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 3 / 34.9Missense obs/exp: 405 / 593.2Syn Z: -0.14
DN
0.6065th %ile
GOF
0.76top 25%
LOF
0.4825th %ile

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFLOEUF 0.22
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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic62
Likely Pathogenic5
VUS130
Likely Benign65
Benign19
Conflicting2
62
Pathogenic
5
Likely Pathogenic
130
VUS
65
Likely Benign
19
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
62
0
62
Likely Pathogenic
0
3
2
0
5
VUS
3
109
18
0
130
Likely Benign
0
31
7
27
65
Benign
0
2
6
11
19
Conflicting
2
Total31459538283

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

View in ClinVar →

Protein Context — Lollipop Plot

ATP2B3 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Primary Aldosteronism.
Funder JW·Hypertension
2019Review
Familial forms and molecular profile of primary hyperaldosteronism.
Araujo-Castro M et al.·Hipertens Riesgo Vasc
2022Review
Top 5 results · since 2015Search PubMed ↗