Alagille Syndrome

Clin Liver Dis. 2018 Nov;22(4):625-641. doi: 10.1016/j.cld.2018.06.001. Epub 2018 Aug 22.

Abstract

Alagille syndrome is a complex multisystem autosomal dominant disorder with a wide variability in penetrance of clinical features. A majority of patients have pathogenic mutations in either the JAG1 gene, encoding a Notch pathway ligand, or the receptor NOTCH2. No genotype-phenotype correlations have been found in any organ system. Liver disease is a major cause of morbidity in this population, whereas cardiac and vascular involvement accounts for most of the mortality. Current therapies are supportive, but the future is promising for the development of targeted interventions to augment Notch pathway signaling in involved tissues.

Keywords: Cholestasis; JAG1; Liver transplant; NOTCH2; Pediatric.

Publication types

  • Review

MeSH terms

  • Alagille Syndrome / complications*
  • Alagille Syndrome / diagnosis
  • Alagille Syndrome / genetics*
  • Alagille Syndrome / therapy
  • Genetic Testing
  • Humans
  • Jagged-1 Protein / genetics
  • Liver Transplantation
  • Receptor, Notch2 / genetics
  • Signal Transduction

Substances

  • JAG1 protein, human
  • Jagged-1 Protein
  • NOTCH2 protein, human
  • Receptor, Notch2