PSMB6

Chr 17

proteasome 20S subunit beta 6

Also known as: DELTA, LMPY

PSMB6 encodes a beta subunit of the 20S proteasome core complex that degrades intracellular proteins through both ubiquitin-dependent and ubiquitin-independent pathways, displaying peptidylglutamyl-hydrolizing activity within the complex. Mutations cause autosomal recessive proteasome-associated autoinflammatory syndrome with chronic atypical neutrophilic dermatosis, characterized by early-onset recurrent fever, distinctive skin lesions, and systemic inflammation. This gene shows tolerance to loss-of-function variants in the general population, consistent with its recessive inheritance pattern.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.83
Clinical SummaryPSMB6
Population Constraint (gnomAD)
Low constraint (pLI 0.02) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.83LOEUF
pLI 0.016
Z-score 1.99
OE 0.40 (0.210.83)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.46Z-score
OE missense 0.66 (0.560.79)
99 obs / 149.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.40 (0.210.83)
00.351.4
Missense OE0.66 (0.560.79)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 5 / 12.6Missense obs/exp: 99 / 149.1Syn Z: -0.26
DN
0.75top 25%
GOF
0.6540th %ile
LOF
0.2777th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PSMB6 · 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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