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Abbonante, V.

Publications and source records attributed to Abbonante, V..

2 recordsLinked to original sources

Fibronectin silencing remodels the fibrotic bone marrow niche

Bone marrow (BM) fibrosis is a defining feature of Primary Myelofibrosis, yet the extracellular matrix (ECM) that sustains the fibrotic niche remains largely untargeted therapeutically. Fibronectin (FN), a key organizer of ECM architecture and growth factor signaling, accumulates in the fibrotic BM niche and correlates with disease severity. Here, we investigated whether systemic FN depletion can disrupt pathological remodeling of the BM niche. We developed a chemically modified antisense oligonucleotide targeting FN1 (FN ASO), which reduced FN expression and assembly and impaired transforming growth factor-{beta}1 (TGF-{beta}1)-induced activation of mesenchymal stromal cells. Systemic FN ASO administration efficiently depleted circulating and BM FN in mice and was well tolerated during short-term treatment. In two complementary models of BM fibrosis, FN silencing reduced FN matrix organization, stromal activation, active TGF{beta}-1, reticulin, and collagen deposition. In Jak2V617F-driven disease, transcriptomic profiling further revealed attenuation of ECM, TGF-{beta}1, and mesenchymal remodeling programs, accompanied by changes in inflammatory signaling. Collectively, these findings identify FN as an active organizer of the fibrotic BM niche and show that its systemic silencing uncouple ECM organization from profibrotic stromal signaling, providing rationale for targeting FN in myelofibrosis with broader therapeutic relevance to fibrosis-driven diseases.

cancer biology↗

Non-neuronal, TGF-β- extracellular matrix restructuring promotes neurodegeneration in a PSP-Richardson syndrome model

Progressive supranuclear palsy-Richardson syndrome (PSP-RS) is a rapidly progressive tauopathy lacking effective therapies. Although tau aggregation is a defining feature, the initiating mechanisms remain elusive. Here we used patient-derived induced pluripotent stem cell midbrain organoids, integrating single-cell transcriptomics, bulk RNA profiling, and quantitative proteomics, to dissect early pathogenic events. We identified vascular leptomeningeal-like cells (VLMCs) as the first altered population, exhibiting TGF-{beta}-driven extracellular matrix (ECM) remodeling enriched in collagens, integrins, and TGFBI. The resulting pathological ECM increased stiffness, induced integrin clustering, and activated RhoA-ROCK-mediated cytoskeletal disorganization. These changes sustained PI3K-AKT and MAPK-ERK signaling, suppressed PP2A, hyperactivated mTOR, and impaired autophagy, culminating in tau hyperphosphorylation and mislocalization. Pharmacological inhibition of TGF-{beta}, AKT, ERK, or mTORC1 restored autophagic flux, reduced tau burden, and rescued neuronal architecture. Our findings establish non-neuronal, matrix-producing niche cells as upstream drivers of tauopathy and reveal TGF-{beta}-mediated ECM restructuring as a mechanochemical trigger of neurodegeneration, opening multiple therapeutic avenues for PSP-RS and related tauopathies.

neuroscience↗