USPL1

Chr 13

ubiquitin specific peptidase like 1

Also known as: C13orf22, D13S106E, bA121O19.1

USPL1 encodes a SUMO-specific isopeptidase that removes SUMO modifications from proteins and plays a key role in RNA polymerase II-mediated snRNA transcription in Cajal bodies. Mutations cause autosomal recessive neurodevelopmental disorder with microcephaly, epilepsy, and brain malformations. The gene shows high constraint against loss-of-function variants (LOEUF 0.456), consistent with its essential role in cellular function.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 0.46
Clinical SummaryUSPL1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.28) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.46LOEUF
pLI 0.022
Z-score 4.24
OE 0.28 (0.170.46)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.43Z-score
OE missense 0.95 (0.881.02)
528 obs / 556.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.28 (0.170.46)
00.351.4
Missense OE0.95 (0.881.02)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 11 / 39.9Missense obs/exp: 528 / 556.4Syn Z: 0.05

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

USPL1 · 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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Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗