TP53RK

Chr 20AR

TP53 regulating kinase

Also known as: BUD32, C20orf64, GAMOS4, Nori-2, Nori-2p, PRPK, TPRKB, dJ101A2

TP53RK is a component of the EKC/KEOPS complex required for threonylcarbamoyl modification of tRNAs and functions as an atypical protein kinase that phosphorylates p53 at serine-15. Mutations cause Galloway-Mowat syndrome 4, a disorder characterized by early-onset nephrotic syndrome and microcephaly with developmental delay. This condition follows autosomal recessive inheritance.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 1.651 OMIM phenotype
Clinical SummaryTP53RK
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

Tolerant — LoF & missense variants common in population
LoF Constraint
1.65LOEUF
pLI 0.001
Z-score 0.39
OE 0.83 (0.431.65)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.20Z-score
OE missense 0.95 (0.821.11)
119 obs / 125.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.83 (0.431.65)
00.351.4
Missense OE0.95 (0.821.11)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 5 / 6.0Missense obs/exp: 119 / 125.2Syn Z: 0.14
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongTP53RK-related Galloway-Mowat syndromeLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.7033th %ile
GOF
0.5072th %ile
LOF
0.3452th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

TP53RK · 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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