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Alvarez-Fischer, D.

Publications and source records attributed to Alvarez-Fischer, D..

2 recordsLinked to original sources

Microglial Foxo3 shapes dopaminergic vulnerability in Parkinson disease

Neurodegenerative diseases such as Parkinsons disease (PD) result from complex interactions between neuronal stress and the surrounding tissue environment, yet the determinants that govern this interplay remain incompletely understood. While neuronal responses to mitochondrial dysfunction and proteotoxic stress have been extensively characterized, the contribution of the microglial state to disease progression remains unclear. Here, we identify the transcription factor Foxo3 as a key regulator of dopaminergic vulnerability acting predominantly through microglia-, rather than neuron-intrinsic, mechanisms. Foxo3 was rapidly induced and translocated to the nucleus in dopaminergic-like cells in response to mitochondrial complex I inhibition and -synuclein aggregation, indicating activation of a conserved neuronal stress response. However, neuron-specific deletion of Foxo3 attenuated early Parkinson-like transcriptional signatures but did not confer sustained in vivo neuroprotection. In contrast, microglia-specific deletion of Foxo3 provided robust and durable protection against dopaminergic degeneration across complementary mouse models, including both MPTP intoxication and -synuclein-driven pathology. Translatomic profiling revealed that Foxo3 deficiency induces extensive transcriptional remodeling of microglia at baseline, establishing a distinct state enriched in immune-related and phagocytic pathways that remains compatible with tissue homeostasis. Strikingly, neuroprotection occurred despite minimal transcriptional reprogramming following neurotoxic insult, suggesting that disease outcome is dictated by the baseline microglial state rather than by the magnitude of transcriptional responses to neurotoxic insult. Mechanistically, neuroprotection correlated with selective modulation of discrete signaling nodes, including reinforcement of the TREM2-TYROBP axis and attenuation of pathways involved in intracellular signal amplification, rather than with broad suppression of inflammatory programs. These findings indicate that Foxo3 does not primarily regulate the magnitude of microglial activation, but instead defines the baseline configuration and response thresholds that shape subsequent neuroimmune dynamics. Together, our results identify baseline microglial state as a determinant of neurodegenerative trajectory and position Foxo3 as a central regulator of this process. More broadly, these findings provide a framework in which microglial state-setting, rather than the magnitude of reactive responses alone, governs dopaminergic vulnerability, with potential implications for therapeutic strategies targeting neuroimmune interactions in Parkinsons disease.

neuroscience↗

Non-cell autonomous OTX2 transcription factor regulates anxiety-related behaviors in the mouse

The Otx2 homeoprotein transcription factor is expressed in the dopaminergic neurons of the ventral tegmental area, a mesencephalic nucleus involved in the control of complex behaviors through its projections to limbic structures, including the ventral hippocampus, amygdala, nucleus accumbens and medial prefrontal cortex. We find adult mice heterozygous for Otx2 show anxiolysis-like phenotype in light-dark box and elevated plus maze paradigms. However, the number of dopaminergic neurons, the integrity of their axons, their projection patterns in target structures, and the amounts of dopamine and dopamine metabolites in targets structures were not modified. Because OTX2 is expressed by the choroid plexus, secreted into cerebrospinal fluid and transferred to parvalbumin interneurons of the cortex, hippocampus, and amygdala, we investigated if this phenotype could result from the decreased synthesis of OTX2 in the choroid plexus. Indeed, the anxiolysis-like phenotype was partially recapitulated in the Otx2+/AA and scFvOtx2tg/0 choroid-plexus dependent non-cell-autonomous OTX2 loss of function mouse models. Furthermore, the phenotype was reversed by the overexpression of Otx2 specifically in choroid plexus of adult Otx2 heterozygous mice. Taken together, OTX2 synthesis by the choroid plexus followed by its secretion into the cerebrospinal fluid is an important regulator of the anxiety phenotype in the mouse.

neuroscience↗