Inherited disorders of bilirubin clearance

Pediatr Res. 2016 Mar;79(3):378-86. doi: 10.1038/pr.2015.247. Epub 2015 Nov 23.

Abstract

Inherited disorders of hyperbilirubinemia may be caused by increased bilirubin production or decreased bilirubin clearance. Reduced hepatic bilirubin clearance can be due to defective (i) unconjugated bilirubin uptake and intrahepatic storage, (ii) conjugation of glucuronic acid to bilirubin (e.g., Gilbert syndrome, Crigler-Najjar syndrome, Lucey-Driscoll syndrome, breast milk jaundice), (iii) bilirubin excretion into bile (Dubin-Johnson syndrome), or (iv) conjugated bilirubin re-uptake (Rotor syndrome). In this review, the molecular mechanisms and clinical manifestations of these conditions are described, as well as current approaches to diagnosis and therapy.

Publication types

  • Research Support, N.I.H., Extramural
  • Review

MeSH terms

  • Animals
  • Bile / chemistry
  • Bilirubin / blood*
  • Bilirubin / chemistry
  • Bilirubin / metabolism
  • Crigler-Najjar Syndrome / genetics
  • Gilbert Disease / genetics
  • Glucuronic Acid / chemistry
  • Glucuronosyltransferase / genetics
  • Humans
  • Hyperbilirubinemia, Hereditary / diagnosis
  • Hyperbilirubinemia, Hereditary / genetics*
  • Hyperbilirubinemia, Neonatal / diagnosis
  • Hyperbilirubinemia, Neonatal / genetics*
  • Jaundice, Chronic Idiopathic / genetics
  • Liver / metabolism

Substances

  • Glucuronic Acid
  • UGT1A1 enzyme
  • Glucuronosyltransferase
  • Bilirubin

Supplementary concepts

  • Hyperbilirubinemia, Transient Familial Neonatal