Alzheimer's disease-causing proline substitutions lead to presenilin 1 aggregation and malfunction

EMBO J. 2015 Nov 12;34(22):2820-39. doi: 10.15252/embj.201592042. Epub 2015 Oct 5.

Abstract

Do different neurodegenerative maladies emanate from the failure of a mutual protein folding mechanism? We have addressed this question by comparing mutational patterns that are linked to the manifestation of distinct neurodegenerative disorders and identified similar neurodegeneration-linked proline substitutions in the prion protein and in presenilin 1 that underlie the development of a prion disorder and of familial Alzheimer's disease (fAD), respectively. These substitutions were found to prevent the endoplasmic reticulum (ER)-resident chaperone, cyclophilin B, from assisting presenilin 1 to fold properly, leading to its aggregation, deposition in the ER, reduction of γ-secretase activity, and impaired mitochondrial distribution and function. Similarly, reduced quantities of the processed, active presenilin 1 were observed in brains of cyclophilin B knockout mice. These discoveries imply that reduced cyclophilin activity contributes to the development of distinct neurodegenerative disorders, propose a novel mechanism for the development of certain fAD cases, and support the emerging theme that this disorder can stem from aberrant presenilin 1 function. This study also points at ER chaperones as targets for the development of counter-neurodegeneration therapies.

Keywords: Alzheimer's disease; cyclophilin B; presenilin 1; proteostasis.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alzheimer Disease / genetics
  • Alzheimer Disease / metabolism*
  • Alzheimer Disease / pathology
  • Amino Acid Substitution*
  • Animals
  • Brain / metabolism*
  • Brain / pathology
  • Cell Line
  • Mice
  • Mice, Knockout
  • Molecular Chaperones / genetics
  • Molecular Chaperones / metabolism
  • Presenilin-1 / genetics
  • Presenilin-1 / metabolism*
  • Proline / genetics
  • Proline / metabolism
  • Protein Aggregation, Pathological / genetics
  • Protein Aggregation, Pathological / metabolism*
  • Protein Aggregation, Pathological / pathology
  • Protein Folding

Substances

  • Molecular Chaperones
  • Presenilin-1
  • presenilin 1, mouse
  • Proline