WNK1

Chr 12ARAD

WNK lysine deficient protein kinase 1

Also known as: HSAN2, HSN2, KDP, PPP1R167, PRKWNK1, PSK, p65

This gene encodes a serine/threonine protein kinase that regulates blood pressure by controlling sodium and chloride ion transport and acts as a molecular crowding sensor mediating cellular volume regulation. Mutations cause pseudohypoaldosteronism type IIC (autosomal dominant) and hereditary sensory and autonomic neuropathy type II (autosomal recessive), affecting renal electrolyte handling and peripheral sensory function respectively. WNK1 is highly constrained against loss-of-function variants (pLI 1.0, LOEUF 0.126), indicating that complete loss of function is likely incompatible with normal development.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismAR/ADLOEUF 0.132 OMIM phenotypes
Clinical SummaryWNK1
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Gene-Disease Validity (ClinGen)
neuropathy, hereditary sensory and autonomic, type 2A · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📖
GeneReview available — WNK1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.13LOEUF
pLI 1.000
Z-score 8.82
OE 0.07 (0.040.13)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
2.16Z-score
OE missense 0.83 (0.790.87)
1043 obs / 1258.9 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.07 (0.040.13)
00.351.4
Missense OE0.83 (0.790.87)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 7 / 104.2Missense obs/exp: 1043 / 1258.9Syn Z: -0.88
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveWNK1-related neuropathy, hereditary sensory and autonomicLOFAR

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

DN
0.2798th %ile
GOF
0.3491th %ile
LOF
0.80top 5%

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.

Literature Evidence

DNDominant-negative mutation, genetic depletion, or chemical inhibition of WNK1 in immature neurons triggered a hyperpolarizing shift in GABA activity by enhancing KCC2-mediated Cl(-) extrusion.PMID:26126716
GOFWNK4 mutations behave as a loss of function for the Na+-Cl- cotransporter and a gain of function when it comes to ROMK and claudins.PMID:15637347

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

WNK1 · 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
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Familial Hyperkalemic Hypertension.
Cornelius RJ et al.·Compr Physiol
2024Review
WNK1 in the kidney.
Bahena-Lopez JP et al.·Curr Opin Nephrol Hypertens
2022Review
Top 5 results · since 2015Search PubMed ↗