Search bioRxiv⌕ Search

Biology subjects

Lo Piccolo, L.

Publications and source records attributed to Lo Piccolo, L..

5 recordsLinked to original sources

Pre-symptomatic proteomic and metabolomic profiling identifies compensated ER-redox-metabolic adaptation and early nuclear vulnerability in neuronal ERO1L toxicity

Aging progressively challenges neuronal proteostasis, redox homeostasis, and metabolism, yet the molecular changes that precede functional decline remain poorly understood. Endoplasmic reticulum oxidoreductin 1 (ERO1), a key regulator of oxidative protein folding, links endoplasmic reticulum (ER) proteostasis with cellular redox balance and is elevated in aging and neurodegenerative contexts. Here, we investigated how neuronal ERO1L elevation reshapes cellular homeostasis before overt dysfunction in Drosophila melanogaster. Endogenous ERO1L expression increased with age, and neuronal ERO1L elevation shortened lifespan and caused progressive locomotor decline. This effect was strongly cell-type dependent, as ERO1L elevation in glia, muscle, or fat body did not produce a comparable survival phenotype. At day 5 post-eclosion, locomotor performance remained preserved and major brain reactive-oxygen-species (ROS) accumulation was not yet detectable, defining a pre-symptomatic stage. Multi-omic profiling at this stage revealed selective remodelling of ER proteostasis, redox defence, and mitochondrial-energy pathways, together with changes in central-carbon, nitrogen, and purine metabolism. In contrast to these broadly adaptive responses, chromatin- and RNA-homeostasis-associated proteins were selectively reduced, accompanied by decreased HP1, dFmr1, and Piwi expression and increased transposable-element transcripts. Thus, neuronal ERO1L elevation establishes a pre-symptomatic state in which proteostatic and metabolic adaptation coexists with early vulnerability of nuclear and RNA-homeostasis pathways, preceding overt oxidative stress and behavioural decline. These findings provide an in vivo framework to investigate how age-associated ERO1L elevation may progressively reduce neuronal resilience during brain aging.

neuroscience↗

A leukemia-derived ENL/AF9 chemical probe enhances neuronal stress resilience and ameliorates ALS phenotypes

Chemical perturbation of chromatin reader proteins provides a precise strategy to interrogate epigenetic control of neuronal stress adaptation. ENL and AF9 are YEATS-domain acyl-lysine readers best characterized in leukemia, but their roles in neurons remain unclear. Here, we use the selective YEATS inhibitor SR-0813 to define ENL/AF9 function in neuronal stress responses across Drosophila and human systems. SR-0813 phenocopies genetic ENL/AF9 reduction by extending lifespan and enhancing stress tolerance in vivo, and improves survival of human neurons under multiple stress conditions, with the strongest effects during endoplasmic reticulum stress. Mechanistically, SR-0813 attenuates PERK-ISR signaling and reduces apoptotic commitment without broadly enhancing proteostasis capacity. Notably, its effects are highly context dependent, conferring protection in stress-signaling-driven models but reduced efficacy or detrimental outcomes under chronic aggregation or mitochondrial stress. These findings identify ENL/AF9 as modulators of stress-response dynamics and highlight YEATS-domain inhibition as a context-dependent strategy to reshape neuronal resilience. GRAPHICAL ABSTRACT O_FIG O_LINKSMALLFIG WIDTH=181 HEIGHT=200 SRC="FIGDIR/small/717610v2_ufig1.gif" ALT="Figure 1"> View larger version (46K): org.highwire.dtl.DTLVardef@e7bee4org.highwire.dtl.DTLVardef@a59d76org.highwire.dtl.DTLVardef@104dd7eorg.highwire.dtl.DTLVardef@69bb51_HPS_FORMAT_FIGEXP M_FIG C_FIG siRNA or the YEATS-domain inhibitor SR-0813 suppress the acyl-lysine reader ENL/AF9, extending lifespan and increasing H2O2 tolerance in Drosophila. In neurons (Drosophila and SH-SY5Y), ENL/AF9 inhibition lowers PERK-ISR signaling and apoptosis while maintaining proteostasis. Effects are context dependent: protective under UPR/ISR-dominant stress but potentially detrimental under aggregation or mitochondrial stress. Created with BioRender.

cell biology↗

Dopaminergic neurons are vulnerable to dysregulation of YEATS2-dependent calcium homeostasis

YEATS2 is a ubiquitously expressed chromatin-associated factor that we recently identified as a novel regulator of dopaminergic (DAergic) synaptic integrity, though its mechanism of action remained unclear. Using Drosophila, we show that neuronal depletion of YEATS2 reshapes the brain transcriptome, marked by downregulation of metabolic genes and upregulation of G protein-coupled receptors (GPCRs). These changes coincide with elevated intracellular calcium, neurobehavioral deficits, and selective DA neuron loss. Importantly, genetic or pharmacological inhibition of the store-operated calcium entry channel Orai restored calcium homeostasis and rescued DA neuron survival. Our findings define a YEATS2-dependent epigenetic-calcium axis that governs DA neuron vulnerability.

neuroscience↗

Diesel exhaust particles induce lasting and age-dependent damage to the brain in Drosophila melanogaster

Diesel exhaust particles (DEP), major air pollutants emitted from automobile engines, contain numerous toxic compounds. While the adverse effects of DEP exposure on the respiratory and cardiovascular systems are well documented, its impact on brain health remains poorly understood. In this study, we employed Drosophila melanogaster as a model organism to investigate the neurological effects of DEP exposure and the impact of exposure cessation across different age groups. Molecular, histopathological, and behavioral markers were assessed before and after exposure to varying doses of DEP at different time intervals.Interestingly, DEP exposure induced age-dependent cellular responses in the brain, including elevated reactive oxygen species (ROS), increased neuroinflammation, and disruption of the blood-brain barrier (BBB). Prolonged exposure led to pronounced vacuolization in the brains of aged flies. While cessation of DEP exposure resulted in partial recovery in young flies particularly when implemented early, aged flies exhibited limited benefit, with persistent evidence of likely irreversible brain damage. Overall, this study invites greater public awareness and careful consideration in public health policy to limit long-term DEP exposure, particularly among older individuals, and to encourage strategies that reduce potential risks to brain health associated with air pollution. Highlights1) DEP exposure is detrimental to the brain. 2) The brain responds to DEP exposure in an age-specific manner. 3) Permanent damage to the brain of old flies results from DEP exposure. 4) Cessation mitigates DEP-induced brain impairments when implemented at a young age. Environmental ImplicationOur study highlights the detrimental, age-dependent effects of diesel exhaust particle (DEP) exposure on brain health, underscoring the urgency of reducing air pollution. The findings support stricter environmental regulations to limit DEP emissions and promote cleaner transportation alternatives. Protecting vulnerable populations, particularly the elderly, from prolonged exposure may help mitigate the long-term neurological impacts of air pollutants and reduce the public health burden.

pharmacology and toxicology↗

Discovery of a pyrazolopyridine alkaloid inhibitor of ERO1A that mitigates neuronal ER stress and age-related decline

Endoplasmic reticulum (ER) stress contributes to the pathogenesis of neurodegenerative and age-associated diseases, motivating the search for compounds that enhance ER-stress resilience. Modulation of ER-redox pathways, including those associated with the oxidase ERO1A, can attenuate maladaptive unfolded protein response (UPR) signaling and improve cellular stress tolerance. Here we developed an integrative discovery strategy to identify natural compounds that mitigate ER-stress-associated phenotypes across cellular and organismal models. Structure-informed virtual screening guided by ERO1A biology prioritized the pyrazolopyridine alkaloid S88. In human SH-SY5Y-derived neurons, S88 improved survival and reduced tunicamycin-induced ER-stress markers. In Drosophila, S88 ameliorated neuromuscular and locomotor phenotypes in a UBQLN2-associated ALS model and improved aging-related outcomes. Biochemical assays did not detect inhibition of ERO1A or radical scavenging activity by S88, indicating that its molecular target remains to be identified. Together, these findings identify S88 as a natural-product scaffold that enhances ER-stress resilience across neuronal and in vivo models. Graphical abstract Structure-informed screening guided by ERO1A prioritized five natural products for functional validation across cellular and Drosophila models. The pyrazolopyridine alkaloid S88 consistently reduced ER-stress markers, improved neuronal survival, rescued locomotor and neuromuscular defects in an ALS model, and ameliorated aging phenotypes. The direct molecular target of S88 remains to be defined. Generated with assistance from the AI-based visualization tool NotebookLM and refined by the authors. O_FIG O_LINKSMALLFIG WIDTH=200 HEIGHT=185 SRC="FIGDIR/small/664722v2_ufig1.gif" ALT="Figure 1"> View larger version (104K): org.highwire.dtl.DTLVardef@8b97c0org.highwire.dtl.DTLVardef@97de54org.highwire.dtl.DTLVardef@857ce6org.highwire.dtl.DTLVardef@1cb1398_HPS_FORMAT_FIGEXP M_FIG C_FIG

neuroscience↗