Interferon-γ causes myogenic cell dysfunction and senescence in immune myopathies

Idiopathic immune myopathies (IIM) represent a heterogeneous group of diseases, in which muscle lesions result from deregulated immune reactions. Typical histological features include myofibre necrosis, leucocyte infiltration and aberrant myofibre major histocompatibility complex (MHC) expression. To investigate the link between MHC expression, inflammation and muscle lesions, muscle biopsies from IIM patients were analysed by transcriptomics. Both anti-synthetase syndrome (ASS) and inclusion body myositis (IBM) displayed the upregulation of interferon-γ (IFN-γ) and senescence signalling pathways. Notably, IFN-γ expression significantly correlated with myofibre atrophy in ASS and IBM muscle biopsies. In addition to MHC-II expression at the myofibre sarcolemma in IBM, we observed a marked overexpression in the muscle stem cell (MuSC) population, suggesting that resident satellite cells respond to IFN-γ in this condition. To examine the link between IFN-γ and muscle atrophy via MuSCs, we implanted an osmotic pump chronically releasing recombinant mouse IFN-γ in wild-type mice subjected to acute muscle injury. Under IFN-γ exposure, post-injury muscle repair was associated with significantly reduced muscle weight and myofibre diameter, while promoting interstitial fibrosis and fat deposition. The mechanism of action of the IFN-γ-induced myofibre atrophy was further investigated in vitro using cultured human MuSCs. IFN-γ stimulation dramatically impaired MuSC proliferation, fusion and promoted cell senescence. Isolated myofibres from IFN-γ-treated wild-type mice displayed a significant decrease of myogenic regulatory factor MyoD expression and cell cycling, suggesting that IFN-γ also prevents MuSC activation. In vitro, ruxolitinib, a commercially available Janus kinase (JAK)1/2 antagonist, blocked IFN-γ-induced expression of MHC-II, restored normal MuSC proliferation and reduced β-galactosidase activity, a marker of cell senescence. In vivo, oral delivery of ruxolitinib improved myofibre size and biomarkers of muscle atrophy. Our study provides multiple lines of evidence that IFN-γ may mediate muscle atrophy in IBM patients. The identified mechanism of action involves JAK1/2 pathways, which impair MuSC function by preventing post-lesion myogenesis and promoting cell senescence. Our data suggest that repurposing JAK1/2 inhibitors may offer a new therapeutic option for treating IBM, a condition known for its resistance to classical immunosuppressant drugs, despite their effectiveness in other IIM.

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Publication Details

Journal
Brain
Published
2025-04-29
DOI
https://doi.org/10.1093/brain/awaf153
Citations
7
Primary Topic
Inflammatory Myopathies and Dermatomyositis
Type
article
Field-Weighted Citation Impact
9.48

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article

Interferon-γ causes myogenic cell dysfunction and senescence in immune myopathies

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7 citations
Brain
Inflammatory Myopathies and Dermatomyositis
9.48
article

Interferon-γ causes myogenic cell dysfunction and senescence in immune myopathies

Edoardo Malfatti, Maximilien Bencze, Juliette Berthier, Sarah Souvannanorath, Baptiste Periou, François‐Jérôme Authier, Romain K. Gherardi, Frédéric Relaix, Yasmine Baba‐Amer, Ludovic Martin, Cyrielle Hou, Marianne Gervais, Emmanuèle Lechapt
article en
7 citations

Abstract

Idiopathic immune myopathies (IIM) represent a heterogeneous group of diseases, in which muscle lesions result from deregulated immune reactions. Typical histological features include myofibre necrosis, leucocyte infiltration and aberrant myofibre major histocompatibility complex (MHC) expression. To investigate the link between MHC expression, inflammation and muscle lesions, muscle biopsies from IIM patients were analysed by transcriptomics. Both anti-synthetase syndrome (ASS) and inclusion body myositis (IBM) displayed the upregulation of interferon-γ (IFN-γ) and senescence signalling pathways. Notably, IFN-γ expression significantly correlated with myofibre atrophy in ASS and IBM muscle biopsies. In addition to MHC-II expression at the myofibre sarcolemma in IBM, we observed a marked overexpression in the muscle stem cell (MuSC) population, suggesting that resident satellite cells respond to IFN-γ in this condition. To examine the link between IFN-γ and muscle atrophy via MuSCs, we implanted an osmotic pump chronically releasing recombinant mouse IFN-γ in wild-type mice subjected to acute muscle injury. Under IFN-γ exposure, post-injury muscle repair was associated with significantly reduced muscle weight and myofibre diameter, while promoting interstitial fibrosis and fat deposition. The mechanism of action of the IFN-γ-induced myofibre atrophy was further investigated in vitro using cultured human MuSCs. IFN-γ stimulation dramatically impaired MuSC proliferation, fusion and promoted cell senescence. Isolated myofibres from IFN-γ-treated wild-type mice displayed a significant decrease of myogenic regulatory factor MyoD expression and cell cycling, suggesting that IFN-γ also prevents MuSC activation. In vitro, ruxolitinib, a commercially available Janus kinase (JAK)1/2 antagonist, blocked IFN-γ-induced expression of MHC-II, restored normal MuSC proliferation and reduced β-galactosidase activity, a marker of cell senescence. In vivo, oral delivery of ruxolitinib improved myofibre size and biomarkers of muscle atrophy. Our study provides multiple lines of evidence that IFN-γ may mediate muscle atrophy in IBM patients. The identified mechanism of action involves JAK1/2 pathways, which impair MuSC function by preventing post-lesion myogenesis and promoting cell senescence. Our data suggest that repurposing JAK1/2 inhibitors may offer a new therapeutic option for treating IBM, a condition known for its resistance to classical immunosuppressant drugs, despite their effectiveness in other IIM.

BrainVol. 148(8)
Inserm (FR), Université Paris-Est Créteil (FR), Laboratoire National de Référence (MA), Hôpitaux Universitaires Henri-Mondor (FR)
French Muscular Dystrophy Association, Agence Nationale de la Recherche
Good health and well-being
Openalex Percentile: Top 2%
Inflammatory Myopathies and Dermatomyositis
9.48
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