PKN1

Chr 19

protein kinase N1

Also known as: DBK, PAK-1, PAK1, PKN, PKN-ALPHA, PRK1, PRKCL1

PKN1 encodes a serine/threonine protein kinase that regulates the actin cytoskeleton and intermediate filaments by phosphorylating proteins including vimentin, neurofilament proteins, and tau, and also functions in transcriptional regulation. Mutations cause autosomal dominant intellectual disability with microcephaly and seizures, typically presenting in early childhood. The gene shows significant constraint against loss-of-function variants (LOEUF = 0.39), consistent with its role in neurodevelopmental disease.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
10
Pubs (1 yr)
14
P/LP submissions
0%
P/LP missense
0.39
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryPKN1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.24) despite low pLI — interpret in context.
📋
ClinVar Variants
14 unique Pathogenic / Likely Pathogenic· 173 VUS of 239 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.39LOEUF
pLI 0.204
Z-score 4.82
OE 0.24 (0.150.39)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.34Z-score
OE missense 0.84 (0.780.91)
496 obs / 587.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.24 (0.150.39)
00.351.4
Missense OE0.84 (0.780.91)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 11 / 46.4Missense obs/exp: 496 / 587.7Syn Z: 0.11
DN
0.6549th %ile
GOF
0.6832th %ile
LOF
0.4135th %ile

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

GOFprediction above median
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

239 submitted variants in ClinVar

Classification Summary

Pathogenic13
Likely Pathogenic1
VUS173
Likely Benign9
Benign4
13
Pathogenic
1
Likely Pathogenic
173
VUS
9
Likely Benign
4
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
13
0
13
Likely Pathogenic
0
0
1
0
1
VUS
0
170
3
0
173
Likely Benign
0
4
2
3
9
Benign
0
1
1
2
4
Total0175205200

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

View in ClinVar →

Protein Context — Lollipop Plot

PKN1 · 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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