EPHB4

Chr 7AD

EPH receptor B4

Also known as: CMAVM2, HFASD, HTK, LMPHM7, MYK1, TYRO11

EPHB4 encodes a receptor tyrosine kinase that binds ephrin-B ligands and controls cellular repulsion, segregation, and migration, playing a central role in vascular development and angiogenesis. Mutations cause autosomal dominant capillary malformation-arteriovenous malformation and lymphatic malformation, affecting vascular and lymphatic system development. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.422), and these vascular malformations can present from infancy through childhood.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.422 OMIM phenotypes
Clinical SummaryEPHB4
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Gene-Disease Validity (ClinGen)
EPHB4-associated vascular malformation spectrum · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.27) despite low pLI — interpret in context.
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.42LOEUF
pLI 0.013
Z-score 4.87
OE 0.27 (0.180.42)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.30Z-score
OE missense 0.74 (0.690.80)
475 obs / 638.2 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.27 (0.180.42)
00.351.4
Missense OE0.74 (0.690.80)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 14 / 51.8Missense obs/exp: 475 / 638.2Syn Z: -1.02
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedEPHB4-related capillary malformation-arteriovenous malformationLOFAD
DN
0.74top 25%
GOF
0.79top 25%
LOF
0.2679th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

EPHB4 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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