CRACR2A

Chr 12

calcium release activated channel regulator 2A

Also known as: EFCAB4B, RAB46

The CRACR2A protein is a calcium-binding protein that regulates store-operated calcium entry in T-cells by controlling the clustering and activation of CRAC channels (ORAI1 and STIM1) at plasma membrane-endoplasmic reticulum junctions. Mutations cause combined immunodeficiency with autoimmunity and hemophagocytic lymphohistiocytosis, inherited in an autosomal recessive pattern. The gene shows extremely strong constraint against loss-of-function variants (pLI near 1), indicating that heterozygous loss-of-function is likely incompatible with normal development.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
4
Pubs (1 yr)
63
P/LP submissions
0%
P/LP missense
0.87
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryCRACR2A
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Gene-Disease Validity (ClinGen)
combined immunodeficiency · ARLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
63 unique Pathogenic / Likely Pathogenic· 123 VUS of 226 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.87LOEUF
pLI 0.000
Z-score 2.22
OE 0.62 (0.450.87)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.93Z-score
OE missense 0.87 (0.800.95)
357 obs / 409.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.62 (0.450.87)
00.351.4
Missense OE0.87 (0.800.95)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 24 / 39.0Missense obs/exp: 357 / 409.9Syn Z: 0.87
DN
0.6746th %ile
GOF
0.7127th %ile
LOF
0.3163th %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

226 submitted variants in ClinVar

Classification Summary

Pathogenic59
Likely Pathogenic4
VUS123
Likely Benign9
Benign19
59
Pathogenic
4
Likely Pathogenic
123
VUS
9
Likely Benign
19
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
59
0
59
Likely Pathogenic
0
0
4
0
4
VUS
0
115
8
0
123
Likely Benign
0
5
3
1
9
Benign
0
5
12
2
19
Total0125863214

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

View in ClinVar →

Protein Context — Lollipop Plot

CRACR2A · 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.

Search ClinicalTrials.gov →
Clinical Literature
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