Progranulin deficiency leads to reduced glucocerebrosidase activity

PLoS One. 2019 Jul 10;14(7):e0212382. doi: 10.1371/journal.pone.0212382. eCollection 2019.

Abstract

Mutation in the GRN gene, encoding the progranulin (PGRN) protein, shows a dose-dependent disease correlation, wherein haploinsufficiency results in frontotemporal lobar degeneration (FTLD) and complete loss results in neuronal ceroid lipofuscinosis (NCL). Although the exact function of PGRN is unknown, it has been increasingly implicated in lysosomal physiology. Here we report that PGRN interacts with the lysosomal enzyme, glucocerebrosidase (GCase), and is essential for proper GCase activity. GCase activity is significantly reduced in tissue lysates from PGRN-deficient mice. This is further evidence that reduced lysosomal hydrolase activity may be a pathological mechanism in cases of GRN-related FTLD and NCL.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cell Line
  • Disease Models, Animal
  • Female
  • Frontotemporal Lobar Degeneration / genetics
  • Frontotemporal Lobar Degeneration / metabolism
  • Glucosylceramidase / genetics
  • Glucosylceramidase / metabolism*
  • HEK293 Cells
  • Haploinsufficiency
  • Humans
  • Lysosomes / metabolism
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Mutation
  • Neuronal Ceroid-Lipofuscinoses / genetics
  • Neuronal Ceroid-Lipofuscinoses / metabolism
  • Progranulins / deficiency*
  • Progranulins / genetics
  • Recombinant Proteins / genetics
  • Recombinant Proteins / metabolism

Substances

  • GRN protein, human
  • Grn protein, mouse
  • Progranulins
  • Recombinant Proteins
  • Glucosylceramidase