ALPK2

Chr 18

alpha kinase 2

Also known as: HAK

ALPK2 encodes a protein serine/threonine kinase that regulates cardiac development and cardiomyocyte differentiation by negatively regulating Wnt/beta-catenin signaling. Mutations cause autosomal recessive cardiomyopathy with features that can include hypertrophic and dilated patterns. The gene shows moderate constraint against loss-of-function variants, and cardiac manifestations typically present in infancy or early childhood.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
9
Pubs (1 yr)
6
P/LP submissions
0%
P/LP missense
0.81
LOEUF
DN
Mechanism· predicted
Clinical SummaryALPK2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
6 unique Pathogenic / Likely Pathogenic· 312 VUS of 500 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.81LOEUF
pLI 0.000
Z-score 2.84
OE 0.64 (0.500.81)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.41Z-score
OE missense 0.97 (0.921.02)
1084 obs / 1122.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.64 (0.500.81)
00.351.4
Missense OE0.97 (0.921.02)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 45 / 70.8Missense obs/exp: 1084 / 1122.2Syn Z: -0.25
DN
0.7230th %ile
GOF
0.6247th %ile
LOF
0.2970th %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

500 submitted variants in ClinVar

Classification Summary

Pathogenic4
Likely Pathogenic2
VUS312
Likely Benign131
Benign16
4
Pathogenic
2
Likely Pathogenic
312
VUS
131
Likely Benign
16
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
4
0
4
Likely Pathogenic
0
0
2
0
2
VUS
0
312
0
0
312
Likely Benign
0
22
3
106
131
Benign
0
13
2
1
16
Total034711107465

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

View in ClinVar →

Protein Context — Lollipop Plot

ALPK2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

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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