EPHA6

Chr 3

EPH receptor A6

Also known as: EHK-2, EHK2, EK12, EPA6, HEK12, PRO57066

EPHA6 encodes a receptor tyrosine kinase that binds ephrin-A ligands to mediate bidirectional cell signaling important for axon guidance and neural development. Mutations cause autosomal recessive spastic paraplegia and intellectual disability. The gene shows significant constraint against loss-of-function variants (LOEUF 0.357), indicating intolerance to disruption.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.36
Clinical SummaryEPHA6
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.67) — some intolerance to loss-of-function variants.
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ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 145 VUS of 171 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.36LOEUF
pLI 0.670
Z-score 5.05
OE 0.21 (0.130.36)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.50Z-score
OE missense 0.83 (0.770.89)
505 obs / 609.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.21 (0.130.36)
00.351.4
Missense OE0.83 (0.770.89)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 10 / 47.5Missense obs/exp: 505 / 609.4Syn Z: -1.11
DN
0.6455th %ile
GOF
0.7127th %ile
LOF
0.4430th %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

171 submitted variants in ClinVar

Classification Summary

Pathogenic7
Likely Pathogenic3
VUS145
Likely Benign3
Benign2
7
Pathogenic
3
Likely Pathogenic
145
VUS
3
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
7
0
7
Likely Pathogenic
0
0
3
0
3
VUS
0
124
21
0
145
Likely Benign
0
2
0
1
3
Benign
0
1
0
1
2
Total0127312160

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

View in ClinVar →

Protein Context — Lollipop Plot

EPHA6 · 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.

Search ClinicalTrials.gov →
Clinical Literature
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