RAG2

Chr 11AR

recombination activating 2

Also known as: RAG-2

The RAG2 protein forms a complex with RAG1 that mediates DNA cleavage during V(D)J recombination, the process that assembles diverse immunoglobulin and T-cell receptor genes in developing B and T lymphocytes. Mutations cause autosomal recessive severe combined immunodeficiency including Omenn syndrome, characterized by absent B cells and severe immune dysfunction often presenting in infancy. RAG2 acts as a chromatin sensor that recruits the RAG complex to specific histone modifications and is essential for generating functional adaptive immune responses.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Combined cellular and humoral immune defects with granulomasMIM #233650
AR
Omenn syndromeMIM #603554
AR
Severe combined immunodeficiency, B cell-negativeMIM #601457
AR
0
Active trials
71
Pubs (1 yr)
189
P/LP submissions
43%
P/LP missense
0.79
LOEUF
LOF
Mechanism· G2P
Clinical SummaryRAG2
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Gene-Disease Validity (ClinGen)
recombinase activating gene 2 deficiency · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.02) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
119 unique Pathogenic / Likely Pathogenic· 196 VUS of 500 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.79LOEUF
pLI 0.020
Z-score 2.10
OE 0.38 (0.200.79)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.20Z-score
OE missense 0.97 (0.871.07)
257 obs / 266.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.38 (0.200.79)
00.351.4
Missense OE0.97 (0.871.07)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 5 / 13.3Missense obs/exp: 257 / 266.0Syn Z: 0.23
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveRAG2-related Omenn syndromeLOFAR

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

DN
0.6259th %ile
GOF
0.5367th %ile
LOF
0.4628th %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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

500 submitted variants in ClinVar

Classification Summary

Pathogenic48
Likely Pathogenic71
VUS196
Likely Benign162
Benign4
Conflicting17
48
Pathogenic
71
Likely Pathogenic
196
VUS
162
Likely Benign
4
Benign
17
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
19
9
20
0
48
Likely Pathogenic
15
42
14
0
71
VUS
2
180
9
5
196
Likely Benign
0
4
2
156
162
Benign
0
1
3
0
4
Conflicting
17
Total3623648161498

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

View in ClinVar →

Protein Context — Lollipop Plot

RAG2 · 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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