ERBB4

Chr 2AD

erb-b2 receptor tyrosine kinase 4

Also known as: ALS19, HER4, p180erbB4

This gene encodes a tyrosine kinase that functions as a cell surface receptor for neuregulins and EGF family members, regulating development of the heart, central nervous system, and mammary gland through control of cell proliferation, differentiation, and migration. Mutations cause autosomal dominant amyotrophic lateral sclerosis 19. The gene is highly constrained against loss-of-function variation (pLI 0.999, LOEUF 0.265), indicating that haploinsufficiency is likely not tolerated in the general population.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismADLOEUF 0.271 OMIM phenotype
Clinical SummaryERBB4
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Gene-Disease Validity (ClinGen)
amyotrophic lateral sclerosis type 19 · ADLimited

Limited evidence — not for standalone diagnostic reporting

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.
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Clinical Trials
4 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — ERBB4
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.27LOEUF
pLI 0.999
Z-score 6.63
OE 0.16 (0.100.27)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.01Z-score
OE missense 0.79 (0.740.85)
576 obs / 728.7 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.16 (0.100.27)
00.351.4
Missense OE0.79 (0.740.85)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 12 / 73.3Missense obs/exp: 576 / 728.7Syn Z: -2.23
DN
0.5673th %ile
GOF
0.74top 25%
LOF
0.50top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function, gain-of-function and dominant-negative). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOF1 literature citation · LOEUF 0.27
GOFprediction above median
DN1 literature citation

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.

Literature Evidence

DNThe predicted mutant protein is likely to function similarly to an engineered dominant-negative ERBB4 (DN-ErbB4), which causes defects in neuronal migration and synaptic neurotransmission.PMID:18369103
LOFThe severe clinical consequences in our patient demonstrate that the haploinsufficiency of ERBB4 is crucial for intellectual and cognitive function.PMID:23633123

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

ERBB4 · 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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