UTP23

Chr 8

UTP23 small subunit processome component

Also known as: C8orf53

UTP23 encodes a protein involved in ribosomal RNA processing and ribosome biogenesis within the nucleolus. Mutations cause autosomal recessive developmental delay with increased adiposity and delayed myelination, typically presenting in infancy with developmental delays and characteristic brain imaging findings. The gene shows moderate tolerance to loss-of-function variants based on population constraint metrics.

ResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 0.90
Clinical SummaryUTP23
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.35) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.90LOEUF
pLI 0.086
Z-score 1.77
OE 0.35 (0.160.90)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.69Z-score
OE missense 0.82 (0.700.97)
100 obs / 121.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.35 (0.160.90)
00.351.4
Missense OE0.82 (0.700.97)
00.61.4
Synonymous OE1.13
01.21.6
LoF obs/exp: 3 / 8.6Missense obs/exp: 100 / 121.4Syn Z: -0.71
DN
0.6550th %ile
GOF
0.5171th %ile
LOF
0.2775th %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

UTP23 · 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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