NOP14

Chr 4

NOP14 nucleolar protein

Also known as: C4orf9, NOL14, RES4-25, RES425, UTP2

The protein is involved in nucleolar processing of pre-18S ribosomal RNA and nuclear export of 40S pre-ribosomal subunits to the cytoplasm. Mutations cause autosomal recessive developmental delay with short stature, dysmorphic features, and intellectual disability. This gene is highly constrained against loss-of-function variants (LOEUF 0.58), consistent with its essential role in ribosome biogenesis.

Summary from RefSeq, UniProt
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0
Active trials
11
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.58
LOEUF
DN
Mechanism· predicted
Clinical SummaryNOP14
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

Moderate LoF intolerance
LoF Constraint
0.58LOEUF
pLI 0.000
Z-score 3.83
OE 0.39 (0.270.58)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-0.84Z-score
OE missense 1.11 (1.031.19)
544 obs / 491.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.39 (0.270.58)
00.351.4
Missense OE1.11 (1.031.19)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 18 / 46.0Missense obs/exp: 544 / 491.9Syn Z: -0.81
DN
0.7035th %ile
GOF
0.4579th %ile
LOF
0.3260th %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

NOP14 · 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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Clinical Literature
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