UTP11

Chr 1

UTP11 small subunit processome component

Also known as: CGI-94, CGI94, UTP11L

UTP11 encodes a protein that is part of the small subunit processome and is essential for ribosomal small subunit biogenesis, specifically involved in nucleolar processing of pre-18S ribosomal RNA. Mutations cause microcephaly with early-onset seizures and severe developmental delay, inherited in an autosomal recessive pattern. The gene shows minimal constraint against loss-of-function variants, which is consistent with the recessive inheritance pattern observed in affected patients.

OMIMResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 1.18
Clinical SummaryUTP11
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
1.18LOEUF
pLI 0.000
Z-score 1.01
OE 0.74 (0.481.18)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.42Z-score
OE missense 0.90 (0.781.04)
129 obs / 143.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.74 (0.481.18)
00.351.4
Missense OE0.90 (0.781.04)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 13 / 17.6Missense obs/exp: 129 / 143.2Syn Z: -0.40
DN
0.7230th %ile
GOF
0.3986th %ile
LOF
0.3162th %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

UTP11 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC