Neurotoxic mechanisms by which the USP14 inhibitor IU1 depletes ubiquitinated proteins and Tau in rat cerebral cortical neurons: Relevance to Alzheimer's disease

Biochim Biophys Acta Mol Basis Dis. 2017 Jun;1863(6):1157-1170. doi: 10.1016/j.bbadis.2017.03.017. Epub 2017 Apr 1.

Abstract

In Alzheimer's disease proteasome activity is reportedly downregulated, thus increasing it could be therapeutically beneficial. The proteasome-associated deubiquitinase USP14 disassembles polyubiquitin-chains, potentially delaying proteasome-dependent protein degradation. We assessed the protective efficacy of inhibiting or downregulating USP14 in rat and mouse (Usp14axJ) neuronal cultures treated with prostaglandin J2 (PGJ2). IU1 concentrations (HIU1>25μM) reported by others to inhibit USP14 and be protective in non-neuronal cells, reduced PGJ2-induced Ub-protein accumulation in neurons. However, HIU1 alone or with PGJ2 is neurotoxic, induces calpain-dependent Tau cleavage, and decreases E1~Ub thioester levels and 26S proteasome assembly, which are energy-dependent processes. We attribute the two latter HIU1 effects to ATP-deficits and mitochondrial Complex I inhibition, as shown herein. These HIU1 effects mimic those of mitochondrial inhibitors in general, thus supporting that ATP-depletion is a major mediator of HIU1-actions. In contrast, low IU1 concentrations (LIU1≤25μM) or USP14 knockdown by siRNA in rat cortical cultures or loss of USP14 in cortical cultures from ataxia (Usp14axJ) mice, failed to prevent PGJ2-induced Ub-protein accumulation. PGJ2 alone induces Ub-protein accumulation and decreases E1~Ub thioester levels. This seemingly paradoxical result may be attributed to PGJ2 inhibiting some deubiquitinases (such as UCH-L1 but not USP14), thus triggering Ub-protein stabilization. Overall, IU1-concentrations that reduce PGJ2-induced accumulation of Ub-proteins are neurotoxic, trigger calpain-mediated Tau cleavage, lower ATP, E1~Ub thioester and E1 protein levels, and reduce proteasome activity. In conclusion, pharmacologically inhibiting (with low or high IU1 concentrations) or genetically down-regulating USP14 fail to enhance proteasomal degradation of Ub-proteins or Tau in neurons.

Keywords: Alzheimer's; Calpain; Deubiquitinase; Mitochondria; Tau; USP14; Ubiquitin-activating enzyme.

Publication types

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

MeSH terms

  • Alzheimer Disease / drug therapy
  • Alzheimer Disease / genetics
  • Alzheimer Disease / metabolism*
  • Alzheimer Disease / pathology
  • Animals
  • Cerebral Cortex / metabolism*
  • Cerebral Cortex / pathology
  • Dose-Response Relationship, Drug
  • Electron Transport Complex I / genetics
  • Electron Transport Complex I / metabolism
  • Mice
  • Mitochondria / genetics
  • Mitochondria / metabolism
  • Mitochondria / pathology
  • Neurons / metabolism*
  • Neurons / pathology
  • Neurotoxicity Syndromes / drug therapy
  • Neurotoxicity Syndromes / genetics
  • Neurotoxicity Syndromes / metabolism*
  • Neurotoxicity Syndromes / pathology
  • Pyrroles / pharmacology*
  • Pyrrolidines / pharmacology*
  • Rats
  • Rats, Sprague-Dawley
  • Ubiquitin Thiolesterase / antagonists & inhibitors*
  • Ubiquitin Thiolesterase / genetics
  • Ubiquitin Thiolesterase / metabolism
  • Ubiquitination / drug effects*
  • tau Proteins / genetics
  • tau Proteins / metabolism*

Substances

  • 1-(1-(4-fluorophenyl)-2,5-dimethylpyrrol-3-yl)-2-pyrrolidin-1-ylethanone
  • Mapt protein, mouse
  • Mapt protein, rat
  • Pyrroles
  • Pyrrolidines
  • USP14 protein, rat
  • Usp14 protein, mouse
  • tau Proteins
  • Ubiquitin Thiolesterase
  • Electron Transport Complex I