USP13

Chr 3

ubiquitin specific peptidase 13

Also known as: ISOT3, IsoT-3

Enables several functions, including BAT3 complex binding activity; peptidase activity; and proteasome binding activity. Involved in several processes, including maintenance of unfolded protein; regulation of DNA-templated transcription; and regulation of catabolic process. Acts upstream of or within protein deubiquitination and protein stabilization. Predicted to be located in nucleoplasm. Predicted to be active in cytosol and nucleus. [provided by Alliance of Genome Resources, Apr 2025]

OMIMResearchGenerating clinical summary…
DNmechanismLOEUF 0.75
Clinical SummaryUSP13
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — 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?
0.75LOEUF
pLI 0.000
Z-score 2.99
OE 0.54 (0.400.75)
Tolerant

Typical tolerance to LoF variation

Missense Constraint?
1.90Z-score
OE missense 0.76 (0.700.83)
376 obs / 494.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.54 (0.400.75)
00.351.4
Missense OE?0.76 (0.700.83)
00.61.4
Synonymous OE?0.93
01.21.6
LoF obs/exp: 27 / 49.7Missense obs/exp: 376 / 494.9Syn Z: 0.75

This gene — mechanism propensity

DN
0.6162th %ile
GOF
0.5465th %ile
LOF
0.2969th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

USP13 · 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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