MSRB2

Chr 10

methionine sulfoxide reductase B2

Also known as: CBS-1, CBS1, CGI-131, MSRB, PILB

The MSRB2 protein is a mitochondrial methionine-sulfoxide reductase that reduces oxidized methionine residues back to methionine and helps preserve mitochondrial integrity during oxidative stress. Mutations cause autosomal recessive sensorineural hearing loss, which can be congenital or early-onset and may be associated with mitochondrial dysfunction. This gene primarily affects the auditory system, though the mitochondrial localization suggests potential for broader cellular effects.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
7
Pubs (1 yr)
9
P/LP submissions
0%
P/LP missense
1.56
LOEUF
Mechanism
Clinical SummaryMSRB2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
9 unique Pathogenic / Likely Pathogenic· 33 VUS of 59 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.56LOEUF
pLI 0.000
Z-score 0.40
OE 0.85 (0.481.56)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.63Z-score
OE missense 0.81 (0.680.99)
75 obs / 92.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.85 (0.481.56)
00.351.4
Missense OE0.81 (0.680.99)
00.61.4
Synonymous OE0.80
01.21.6
LoF obs/exp: 7 / 8.3Missense obs/exp: 75 / 92.2Syn Z: 0.97

ClinVar Variant Classifications

59 submitted variants in ClinVar

Classification Summary

Pathogenic8
Likely Pathogenic1
VUS33
Likely Benign6
Benign1
8
Pathogenic
1
Likely Pathogenic
33
VUS
6
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
8
0
8
Likely Pathogenic
0
0
1
0
1
VUS
0
33
0
0
33
Likely Benign
0
5
0
1
6
Benign
0
0
1
0
1
Total03810149

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

View in ClinVar →

Protein Context — Lollipop Plot

MSRB2 · 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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