Congenital jaundice in rats with a mutation in a multidrug resistance-associated protein gene

Science. 1996 Feb 23;271(5252):1126-8. doi: 10.1126/science.271.5252.1126.

Abstract

The human Dubin-Johnson syndrome and its animal model, the TR(-) rat, are characterized by a chronic conjugated hyperbilirubinemia. TR(-) rats are defective in the canalicular multispecific organic anion transporter (cMOAT), which mediates hepatobiliary excretion of numerous organic anions. The complementary DNA for rat cmoat, a homolog of the human multidrug resistance gene (hMRP1), was isolated and shown to be expressed in the canalicular membrane of hepatocytes. In the TR(-) rat, a single-nucleotide deletion in this gene resulted in a reduced messenger RNA level and absence of the protein. It is likely that this mutation accounts for the TR(-) phenotype.

MeSH terms

  • ATP-Binding Cassette Transporters / analysis
  • ATP-Binding Cassette Transporters / chemistry
  • ATP-Binding Cassette Transporters / genetics*
  • Amino Acid Sequence
  • Animals
  • Anion Transport Proteins
  • Base Sequence
  • Carrier Proteins / analysis
  • Carrier Proteins / chemistry
  • Carrier Proteins / genetics*
  • Cell Membrane / chemistry
  • DNA, Complementary / genetics
  • Frameshift Mutation
  • Humans
  • Hyperbilirubinemia, Hereditary / genetics*
  • Liver / chemistry*
  • Liver / cytology
  • Molecular Sequence Data
  • Molecular Weight
  • Multidrug Resistance-Associated Proteins
  • Phenotype
  • Rats
  • Rats, Wistar
  • Sequence Alignment
  • Sequence Deletion

Substances

  • ATP-Binding Cassette Transporters
  • Anion Transport Proteins
  • Carrier Proteins
  • DNA, Complementary
  • Multidrug Resistance-Associated Proteins

Associated data

  • GENBANK/L49379