GNL2

Chr 1

G protein nucleolar 2

Also known as: HUMAUANTIG, NGP1, Ngp-1, Nog2, Nug2

The protein functions as a GTPase that associates with pre-60S ribosomal subunits in the nucleolus and is required for their nuclear export and maturation. Mutations cause autosomal recessive neurodevelopmental disorder with dysmorphic facies and distal limb anomalies, typically presenting in early childhood with intellectual disability, developmental delays, and characteristic facial features. This gene is highly constrained against loss-of-function variants, indicating that complete loss of protein function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 0.55
Clinical SummaryGNL2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
5 unique Pathogenic / Likely Pathogenic· 108 VUS of 134 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.000
Z-score 3.85
OE 0.36 (0.240.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.28Z-score
OE missense 0.96 (0.891.04)
394 obs / 409.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.36 (0.240.55)
00.351.4
Missense OE0.96 (0.891.04)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 15 / 41.9Missense obs/exp: 394 / 409.7Syn Z: -0.09
DN
0.7326th %ile
GOF
0.5071th %ile
LOF
0.3453th %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

134 submitted variants in ClinVar

Classification Summary

Pathogenic4
Likely Pathogenic1
VUS108
Likely Benign6
4
Pathogenic
1
Likely Pathogenic
108
VUS
6
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
4
0
4
Likely Pathogenic
0
0
1
0
1
VUS
0
101
7
0
108
Likely Benign
0
4
1
1
6
Benign
0
0
0
0
0
Total0105131119

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

View in ClinVar →

Protein Context — Lollipop Plot

GNL2 · 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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