GTPBP1

Chr 22AR

GTP binding protein 1

Also known as: GP-1, GP1, HSPC018, NEDFET1

GTPBP1 encodes a GTPase that delivers aminoacyl-tRNAs to ribosomes during protein synthesis and promotes degradation of faulty mRNAs through RNA quality control mechanisms. Autosomal recessive mutations cause neurodevelopmental disorder with characteristic facial and ectodermal features and tetraparesis. The gene is highly constrained against loss-of-function variants, indicating that functional GTPBP1 protein is essential for normal cellular processes.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.351 OMIM phenotype
Clinical SummaryGTPBP1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.93). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.35LOEUF
pLI 0.930
Z-score 4.01
OE 0.15 (0.070.35)
Highly constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
3.36Z-score
OE missense 0.52 (0.470.59)
205 obs / 392.1 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.15 (0.070.35)
00.351.4
Missense OE0.52 (0.470.59)
00.61.4
Synonymous OE0.86
01.21.6
LoF obs/exp: 4 / 26.1Missense obs/exp: 205 / 392.1Syn Z: 1.34

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

GTPBP1 · 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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Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC