JAG1

Chr 20AD

jagged canonical Notch ligand 1

Also known as: AGS, AGS1, AHD, AWS, CD339, CMT2HH, DCHE, HJ1

The jagged 1 protein functions as a ligand for Notch receptors, mediating Notch signaling pathways involved in hematopoiesis, cardiovascular development, and cell-fate decisions. Mutations cause Alagille syndrome 1 (characterized by liver, heart, eye, and skeletal abnormalities), as well as congenital heart defects, deafness with posterior embryotoxon, tetralogy of Fallot, and axonal Charcot-Marie-Tooth disease type 2HH. JAG1 follows autosomal dominant inheritance and is highly constrained against loss-of-function variants (pLI >0.99).

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.154 OMIM phenotypes
Clinical SummaryJAG1
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
3 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.15LOEUF
pLI 1.000
Z-score 6.88
OE 0.06 (0.030.15)
Highly constrained

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

Missense Constraint
3.25Z-score
OE missense 0.65 (0.600.71)
455 obs / 696.0 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.06 (0.030.15)
00.351.4
Missense OE0.65 (0.600.71)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 4 / 62.8Missense obs/exp: 455 / 696.0Syn Z: 0.13
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveJAG1-related Alagille syndromeLOFAD
DN
0.3395th %ile
GOF
0.4678th %ile
LOF
0.70top 10%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function, gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to loss-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFprediction above median · 1 literature citation · LOEUF 0.15
GOF1 literature citation
DN1 literature citation

Literature Evidence

DNAltogether, our results favor a dominant-negative mechanism of some JAGGED1 mutations in AGS.PMID:17720887
GOFThese data support either a relative loss-of-function or a gain-of-function pathogenetic mechanism in this family and suggest that JAG1 mutations may contribute significantly to common variants of right heart obstructive disease.PMID:11152664
LOFThe majority of JAG1 mutations seen in Alagille syndrome patients are null alleles, suggesting JAG1 haploinsufficiency as a primary cause of this disorder.PMID:11861489

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

JAG1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗