The human peroxisomal multifunctional protein involved in bile acid synthesis: activity measurement, deficiency in Zellweger syndrome and chromosome mapping

Biochim Biophys Acta. 1997 May 24;1360(3):229-40. doi: 10.1016/s0925-4439(97)00003-3.

Abstract

The dehydrogenation of 24R,25R-varanoyl-CoA, the physiological intermediate formed during the peroxisomal breakdown of the bile acid intermediate trihydroxycoprostanic acid, was studied in human liver. The reaction appeared to be catalyzed by two different enzymes. A first one, present in the cytosol, did not discriminate between the four possible varanoyl-CoA isomers and did not require the CoA moiety. The second enzymic activity was associated with peroxisomes and acted only on the 24R,25R-isomer, in which the 24-hydroxy group possesses the D-configuration. The D-specific dehydrogenase is part of a 79 kDa protein which represents the human counterpart of a recently discovered second multifunctional protein in rat liver peroxisomes, named multifunctional protein 2 (MFP-2). Human MFP-2, like its rat counterpart, is also responsible for the formation (by hydratation) of 24R,25R-varanoyl-CoA. A deficiency of MFP-2 in Zellweger liver could be demonstrated immunologically by using antibodies against the rat enzyme and enzymically -- after removal of the cytosol -- by using 24R,25R-varanoyl-CoA. The gene coding for MFP-2 was mapped to chromosome 5q2.3.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • 3-Hydroxyacyl CoA Dehydrogenases*
  • Animals
  • Base Sequence
  • Bile Acids and Salts / biosynthesis*
  • Chromatography, Ion Exchange
  • Chromosome Mapping
  • Enoyl-CoA Hydratase*
  • Humans
  • Isomerases*
  • Liver / metabolism*
  • Molecular Sequence Data
  • Multienzyme Complexes / deficiency
  • Multienzyme Complexes / genetics
  • Multienzyme Complexes / metabolism*
  • Peroxisomal Bifunctional Enzyme
  • Rats
  • Stereoisomerism
  • Subcellular Fractions / metabolism
  • Zellweger Syndrome / genetics
  • Zellweger Syndrome / metabolism*

Substances

  • Bile Acids and Salts
  • Multienzyme Complexes
  • 3-Hydroxyacyl CoA Dehydrogenases
  • EHHADH protein, human
  • Enoyl-CoA Hydratase
  • Peroxisomal Bifunctional Enzyme
  • Isomerases

Associated data

  • GENBANK/Y11393