Biogenesis and Function of Peroxisomes in Human Disease with a Focus on the ABC Transporter

Biol Pharm Bull. 2019;42(5):649-665. doi: 10.1248/bpb.b18-00723.

Abstract

Peroxisomes are indispensable organelles in mammals including humans. They are involved in the β-oxidation of very long chain fatty acids, and the synthesis of ether phospholipids and bile acids. Pre-peroxisomes bud from endoplasmic reticulum and peroxisomal membrane and matrix proteins are imported to the pre-peroxisomes. Then, matured peroxisomes grow by division. Impairment of the biogenesis and function of peroxisomes results in severe diseases. Since I first undertook peroxisome research in Prof. de Duve's laboratory at Rockefeller University in 1985, I have continuously studied peroxisomes for more than 30 years, with a particular focus on the ATP-binding cassette (ABC) transporters. Here, I review the history of peroxisome research, the biogenesis and function of peroxisomes, and peroxisome disease including X-linked adrenoleukodystrophy. The review includes the targeting and function of the ABC transporter subfamily D.

Keywords: ATP-binding cassette (ABC) transporter; X-linked adrenoleukodystrophy; biogenesis; fatty acid metabolism; peroxisome; peroxisome disease.

Publication types

  • Review

MeSH terms

  • ATP-Binding Cassette Transporters / metabolism*
  • Adrenoleukodystrophy / metabolism
  • Animals
  • Humans
  • Peroxisomes / metabolism*

Substances

  • ATP-Binding Cassette Transporters