Loss of Mitochondrial Tusc2/Fus1 Triggers a Brain Pro-Inflammatory Microenvironment and Early Spatial Memory Impairment

Int J Mol Sci. 2024 Jul 5;25(13):7406. doi: 10.3390/ijms25137406.

Abstract

Brain pathological changes impair cognition early in disease etiology. There is an urgent need to understand aging-linked mechanisms of early memory loss to develop therapeutic strategies and prevent the development of cognitive impairment. Tusc2 is a mitochondrial-resident protein regulating Ca2+ fluxes to and from mitochondria impacting overall health. We previously reported that Tusc2-/- female mice develop chronic inflammation and age prematurely, causing age- and sex-dependent spatial memory deficits at 5 months old. Therefore, we investigated Tusc2-dependent mechanisms of memory impairment in 4-month-old mice, comparing changes in resident and brain-infiltrating immune cells. Interestingly, Tusc2-/- female mice demonstrated a pro-inflammatory increase in astrocytes, expression of IFN-γ in CD4+ T cells and Granzyme-B in CD8+T cells. We also found fewer FOXP3+ T-regulatory cells and Ly49G+ NK and Ly49G+ NKT cells in female Tusc2-/- brains, suggesting a dampened anti-inflammatory response. Moreover, Tusc2-/- hippocampi exhibited Tusc2- and sex-specific protein changes associated with brain plasticity, including mTOR activation, and Calbindin and CamKII dysregulation affecting intracellular Ca2+ dynamics. Overall, the data suggest that dysregulation of Ca2+-dependent processes and a heightened pro-inflammatory brain microenvironment in Tusc2-/- mice could underlie cognitive impairment. Thus, strategies to modulate the mitochondrial Tusc2- and Ca2+- signaling pathways in the brain should be explored to improve cognitive health.

Keywords: Fus1; Tusc2; aging; brain immune populations; calcium; cognitive impairment; mitochondria; neuroinflammation; sex-dependent changes.

MeSH terms

  • Animals
  • Astrocytes / metabolism
  • Astrocytes / pathology
  • Brain / metabolism
  • Brain / pathology
  • Cellular Microenvironment
  • Female
  • Hippocampus / metabolism
  • Hippocampus / pathology
  • Inflammation / metabolism
  • Inflammation / pathology
  • Male
  • Membrane Proteins / genetics
  • Membrane Proteins / metabolism
  • Memory Disorders / genetics
  • Memory Disorders / metabolism
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Mitochondria* / metabolism
  • Mitochondrial Proteins / genetics
  • Mitochondrial Proteins / metabolism
  • Spatial Memory*

Substances

  • Membrane Proteins
  • Mitochondrial Proteins
  • Tusc2 protein, mouse

Grants and funding

This work was supported by funds from the following National Institutes of Health (NIH) grants: U54 CA163069, U54 MD007593 and SCI CA182843. TF is supported by the NIH RISE training grant 5R25GM059994. This research was, in part, supported or funded by the NIH AIM-AHEAD grant, Agreement NO. 1OT2OD032581, the NHGRI Diversity Center for Genome Research grant UG3HG013248, the U.S. Department of Energy Grant DE-EM0005266, the American Cancer Society grant DICRIDG-21-071-01, and Chan Zuckerberg Initiative grant under award number CZIF2022-007043. This work was partly accomplished through the Meharry RCMI Research Ca-pacity Core funding U54MD007586. Behavioral studies undertaken in the Vanderbilt Mouse Neurobehavior Core were supported by the NICHD of the NIH Award P50HD103537. The views and conclusions contained in this document are those of the authors and should not be interpreted as representing the official policies, either expressed or implied, of the NIH.