Characterization of a mutant form of human apolipoprotein B (Thr26_Tyr27del) associated with familial hypobetalipoproteinemia

Biochim Biophys Acta. 2016 Apr;1861(4):371-9. doi: 10.1016/j.bbalip.2016.01.014. Epub 2016 Jan 26.

Abstract

We have previously identified a deletion mutant of human apoB [apoB (Thr26_Tyr27del)] in a subject with primary hypobetalipoproteinemia. The present study determined the effect of Thr26_Tyr27del mutation on apoB secretion using transfected McA-RH7777 cells. Transient or stable transfection of apoB-48 containing the Thr26_Tyr27del mutation showed drastically reduced secretion of the mutant as compared to wild-type apoB-48. No lipoproteins containing the mutant apoB-48 were secreted into the medium. Incubation of transfected cells in a lipid-rich medium in the presence of cycloheximide showed rapid turnover of cell-associated mutant apoB-48 as compared to that of wild-type apoB-48. Immunofluorescence experiments showed that the mutant apoB-48 was mostly localized in the endoplasmic reticulum. Treatment with the proteasomal inhibitor MG132 markedly attenuated the turnover of cell-associated mutant apoB-48, whereas treatment with inhibitors of autophagosomal/lysosomal function (e.g. 3-MA or ammonium chloride) had no effect. Taken together, these results indicated that the defective secretion of the Thr26_Tyr27del mutant was associated with increased intracellular degradation of apoB through the proteasome-dependent pathway.

Keywords: Apolipoprotein B mutation; Apolipoprotein B-48 secretion; Hypobetalipoproteinemia; Proteasomal degradation.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Apolipoprotein B-100 / genetics*
  • Apolipoprotein B-100 / metabolism
  • Apolipoprotein B-48 / genetics*
  • Apolipoprotein B-48 / metabolism
  • Cell Line
  • DNA Mutational Analysis
  • Endoplasmic Reticulum / metabolism
  • Genetic Predisposition to Disease
  • Heterozygote
  • Humans
  • Hypobetalipoproteinemias / genetics*
  • Hypobetalipoproteinemias / metabolism
  • Phenotype
  • Proteasome Endopeptidase Complex / metabolism
  • Proteasome Inhibitors / pharmacology
  • Proteolysis
  • Sequence Deletion*
  • Time Factors
  • Transfection

Substances

  • APOB protein, human
  • Apolipoprotein B-100
  • Apolipoprotein B-48
  • Proteasome Inhibitors
  • Proteasome Endopeptidase Complex