NOL12

Chr 22

nucleolar protein 12

Also known as: Nop25, VITO, dJ37E16.7

NOL12 encodes a multifunctional RNA binding protein that regulates nucleolar organization and size, participates in DNA stress response and genome integrity maintenance, and controls levels of key ribosomal proteins fibrillarin and nucleolin. Mutations cause autosomal recessive developmental delay with seizures and microcephaly. The gene shows tolerance to loss-of-function variants (pLI 0.001, LOEUF 1.13), consistent with the recessive inheritance pattern observed clinically.

OMIMResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 1.13
Clinical SummaryNOL12
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
19 unique Pathogenic / Likely Pathogenic· 43 VUS of 77 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.13LOEUF
pLI 0.001
Z-score 1.28
OE 0.57 (0.311.13)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.26Z-score
OE missense 0.94 (0.811.08)
129 obs / 137.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.57 (0.311.13)
00.351.4
Missense OE0.94 (0.811.08)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 6 / 10.5Missense obs/exp: 129 / 137.6Syn Z: -0.44
DN
0.76top 25%
GOF
0.5759th %ile
LOF
0.2968th %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

77 submitted variants in ClinVar

Classification Summary

Pathogenic18
Likely Pathogenic1
VUS43
Likely Benign2
Benign1
Conflicting1
18
Pathogenic
1
Likely Pathogenic
43
VUS
2
Likely Benign
1
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
18
0
18
Likely Pathogenic
0
0
1
0
1
VUS
0
41
2
0
43
Likely Benign
0
2
0
0
2
Benign
0
1
0
0
1
Conflicting
1
Total04421066

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

NOL12 · 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 →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 2 results · since 2015Search PubMed ↗