CDK20

Chr 9

cyclin dependent kinase 20

Also known as: CCRK, CDCH, P42, PNQALRE

CDK20 encodes a cyclin-dependent kinase that controls primary cilium structure and is required for high-level Sonic hedgehog signaling responses in the developing neural tube, and also activates CDK2 to regulate cell cycle progression. Mutations cause autosomal recessive primary microcephaly with seizures and intellectual disability, representing a ciliopathy affecting brain development. This gene is not highly constrained against loss-of-function variants.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.65
Clinical SummaryCDK20
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 0.000
Z-score -0.51
OE 1.13 (0.791.65)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.94Z-score
OE missense 1.18 (1.071.31)
246 obs / 207.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.13 (0.791.65)
00.351.4
Missense OE1.18 (1.071.31)
00.61.4
Synonymous OE1.19
01.21.6
LoF obs/exp: 19 / 16.8Missense obs/exp: 246 / 207.9Syn Z: -1.38
DN
0.6648th %ile
GOF
0.6444th %ile
LOF
0.3647th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction 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

CDK20 · 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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