Duchenne muscular dystrophy (DMD) arises from dystrophin deficiency, a crucial component of the dystrophin-glycoprotein complex (DGC) essential for maintaining cellular structural integrity by linking intracellular actin filaments to the basal lamina. Dysfunctions within this complex, coupled with increased inflammatory immune cell infiltration, contribute to the onset of dilated cardiomyopathy (DCM). This cardiac condition, characterized by necrosis and fibrosis, significantly impairs left ventricular function. Despite various treatment approaches, reliable effects on these pathogenic mechanisms remain elusive. RNA-binding proteins play pivotal roles in modulating pathways often dysregulated in cardiac pathology. Notably, HuR, which is upregulated in fibrotic cardiac regions and modulates innate immune system activation, emerges as a promising target. We investigated HuR expression in cardiac tissues of mdx murine model of DMD and assessed the impact of its inhibition with regards to DCM progression. Our findings reveal that HuR is indeed upregulated in mdx mice, and its inhibition leads to attenuation of cardiac fibrosis and improvement in heart function. These preclinical results underscore the potential of targeting HuR for therapeutic intervention to mitigate DCM-associated pathological changes, warranting further exploration for the development of effective treatments.

Inhibition of HuR/ELAVL-1 attenuates fibrotic progression in Mdx mice with dilated cardiomyopathy / A. Farini, M. Molinaro, D. Mostosi, M. Camera, M. Russo, E. Leonetti, M. Meregalli, L. Prandi, C. Liaci, A. Ghigo, E. Hirsch, G. Merlo, Y. Torrente. - In: CELLULAR AND MOLECULAR LIFE SCIENCES. - ISSN 1420-682X. - 83:1(2026), pp. 49.1-49.22. [10.1007/s00018-025-05979-0]

Inhibition of HuR/ELAVL-1 attenuates fibrotic progression in Mdx mice with dilated cardiomyopathy

D. Mostosi;L. Prandi;Y. Torrente
Ultimo
2026

Abstract

Duchenne muscular dystrophy (DMD) arises from dystrophin deficiency, a crucial component of the dystrophin-glycoprotein complex (DGC) essential for maintaining cellular structural integrity by linking intracellular actin filaments to the basal lamina. Dysfunctions within this complex, coupled with increased inflammatory immune cell infiltration, contribute to the onset of dilated cardiomyopathy (DCM). This cardiac condition, characterized by necrosis and fibrosis, significantly impairs left ventricular function. Despite various treatment approaches, reliable effects on these pathogenic mechanisms remain elusive. RNA-binding proteins play pivotal roles in modulating pathways often dysregulated in cardiac pathology. Notably, HuR, which is upregulated in fibrotic cardiac regions and modulates innate immune system activation, emerges as a promising target. We investigated HuR expression in cardiac tissues of mdx murine model of DMD and assessed the impact of its inhibition with regards to DCM progression. Our findings reveal that HuR is indeed upregulated in mdx mice, and its inhibition leads to attenuation of cardiac fibrosis and improvement in heart function. These preclinical results underscore the potential of targeting HuR for therapeutic intervention to mitigate DCM-associated pathological changes, warranting further exploration for the development of effective treatments.
Dilated cardiomyopathy; Duchenne muscular dystrophy; HuR; RNA binding proteins;
Settore MEDS-12/A - Neurologia
   Assegnazione Dipartimenti di Eccellenza 2023-2027 - Dipartimento di BIOTECNOLOGIE MEDICHE E MEDICINA TRASLAZIONALE
   DECC23_003
   MINISTERO DELL'UNIVERSITA' E DELLA RICERCA

   Multiomics pRofiling of patient spEcific Models to predict druggable targets in severe neuromuscular rare diseases (REMODEL)
   REMODEL
   FONDAZIONE REGIONALE PER LA RICERCA BIOMEDICA

   At the origin of congenital muscular dystrophy: shedding light on the Tdark proteins DPM2 and DPM3
   FONDAZIONE CARIPLO
   GJC21084

   National Center for Gene Therapy and Drugs based on RNA Technology (CN3 RNA)
   CN3 RNA
   MINISTERO DELL'UNIVERSITA' E DELLA RICERCA
   CN00000041

   Generation Of Suitable For Beta-Sarcoglycan Delivery and Targeting Of Cardiac And Skeletal Muscle As Alternative Therapy Of LGMD2E
   GRUPPO FAMILIARI BETA-SARCOGLICANOPATIE ONLUS
   5034919
2026
30-dic-2025
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/2434/1247024
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