bioRxiv Science⌕ Search

Biology subjects

Casali, B. T.

Publications and source records attributed to Casali, B. T..

2 recordsLinked to original sources

Therapeutic targeting of immunometabolism in Alzheimers disease reveals a critical reliance on Hexokinase 2 dosage on microglial activation and disease progression.

Microgliosis and neuroinflammation are prominent features of Alzheimers disease (AD). Disease-responsive microglia meet their increased energy demand by reprogramming metabolism, specifically, switching to favor glycolysis over oxidative phosphorylation. Thus, targeting of microglial immunometabolism might be of therapeutic benefit for treating AD, providing novel and often well understood immune pathways and their newly recognized actions in AD. We report that in the brains of 5xFAD mice and postmortem brains of AD patients, we found a significant increase in the levels of Hexokinase 2 (HK2), an enzyme that supports inflammatory responses by rapidly increasing glycolysis. Moreover, binding of HK2 to mitochondria has been reported to regulate inflammation by preventing mitochondrial dysfunction and NLRP3 inflammasome activation, suggesting that its inflammatory role extends beyond its glycolytic activity. Here we report, that HK2 antagonism selectively affects microglial phenotypes and disease progression in a gene-dose dependent manner. Paradoxically, complete loss of HK2 fails to improve AD progression by exacerbating inflammasome activity while its haploinsufficiency results in reduced pathology and improved cognition in the 5XFAD mice. We propose that the partial antagonism of HK2, is effective in slowed disease progression and inflammation through a non-metabolic mechanism associated with the modulation of NFK{beta} signaling, through its cytosolic target IKB. The complete loss of HK2 affects additional inflammatory mechanisms associated to mitochondrial dysfunction. HighlightsO_LIHexokinase 2, the first and rate-limiting enzyme of glycolysis, is specifically upregulated in plaque-associated microglia of AD mice models and in the postmortem cortex of human AD patients. C_LIO_LIMicroglia haploinsufficient in HK2 exhibit reduced amyloid burden and inflammation as well as improved cognition in a mouse model of AD. Paradoxically, the complete loss of HK2 results in opposite effects, by exacerbating inflammation. C_LIO_LILonidamine, an anticancer drug that inhibits HK2, mimics the salutary effects of HK2 haploinsufficiency in the 5xFAD mice, but only in males during the early stages of disease. C_LIO_LIHK2 deletion induced mitochondrial dysfunction associated to increased expression of inflammasome elements and IL-1{beta}. C_LIO_LIHK2 partial antagonism exerts beneficial effects independent of its energetic or mitochondrial role, likely through cytosolic stabilization of I{kappa}B and inhibition of the NF-{kappa}B pathway, leading to reduced proinflammatory gene expression. C_LI

neuroscience↗

INPP5D expression is associated with risk for Alzheimer's disease and induced by plaque-associated microglia

BackgroundAlzheimers disease (AD) is a progressive neurodegenerative disorder characterized by cognitive decline, robust microgliosis, neuroinflammation, and neuronal loss. Genome-wide association studies recently highlighted a prominent role for microglia in late-onset AD (LOAD). Specifically, inositol polyphosphate-5-phosphatase (INPP5D), also known as SHIP1, is selectively expressed in brain microglia and has been reported to be associated with LOAD. Although INPP5D is likely a crucial player in AD pathophysiology, its role in disease onset and progression remains unclear. MethodsWe performed differential gene expression analysis to investigate INPP5D expression in LOAD and its association with plaque density and microglial markers using transcriptomic (RNA-Seq) data from the Accelerating Medicines Partnership for Alzheimers Disease (AMP-AD) cohort. We also performed quantitative real-time PCR, immunoblotting, and immunofluorescence assays to assess INPP5D expression in the 5xFAD amyloid mouse model. ResultsDifferential gene expression analysis found that INPP5D expression was upregulated in LOAD and positively correlated with amyloid plaque density. In addition, in 5xFAD mice, Inpp5d expression increased as the disease progressed, and selectively in plaque-associated microglia. Increased Inpp5d expression levels in 5xFAD mice were abolished entirely by depleting microglia with the colony-stimulating factor receptor-1 antagonist PLX5622. ConclusionsOur findings show that INPP5D expression increases as AD progresses, predominantly in plaque-associated microglia. Importantly, we provide the first evidence that increased INPP5D expression might be a risk factor in AD, highlighting INPP5D as a potential therapeutic target. Moreover, we have shown that the 5xFAD mouse model is appropriate for studying INPP5D in AD.

neuroscience↗