ArticleMolecular therapy : the journal of the American Society of Gene Therapy2023
A dysfunctional miR-1-TRPS1-MYOG axis drives ERMS by suppressing terminal myogenic differentiation.
Article in Molecular therapy : the journal of the American Society of Gene Therapy, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 10 citations in OpenAlex.
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- Global MyoG research 2004-2024: a bibliometric analysis of trends and translational implications.Experimental biology and medicine (Maywood, N.J.) · 2026Review
- Chinese leek-derived extracellular vesicles ameliorate sarcopenia by regulating mitochondrial biogenesis and autophagy via AMPK and maintaining myosin homeostasis.Journal of nanobiotechnology · 2025Article
- Off-label Use of Dinutuximab Beta in Combination With Chemotherapy in Patients With Ewing Sarcoma: A Retrospective Single-center Case Series.Journal of pediatric hematology/oncology · 2025Article
- Maternal Western-style diet has a persistent effect on offspring gene expression in skeletal muscle of Japanese macaques.Scientific reports · 2025Article
- Emerging role and function of Hippo-YAP/TAZ signaling pathway in musculoskeletal disorders.Stem cell research & therapy · 2024Review
- We need to talk-how muscle stem cells communicate.Frontiers in cell and developmental biology · 2024Review
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Authors and funding
7 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Rhabdomyosarcoma is the most common pediatric soft tissue tumor, comprising two major subtypes: the PAX3/7-FOXO1 fusion-negative embryonal and the PAX3/7-FOXO1 fusion-positive alveolar subtype. Here, we demonstrate that the expression levels of the transcriptional repressor TRPS1 are specifically enhanced in the embryonal subtype, resulting in impaired terminal myogenic differentiation and tumor growth. During normal myogenesis, expression levels of TRPS1 have to decrease to allow myogenic progression, as demonstrated by overexpression of TRPS1 in myoblasts impairing myotube formation. Consequentially, myogenic differentiation in embryonal rhabdomyosarcoma in vitro as well as in vivo can be achieved by reducing TRPS1 levels. Furthermore, we show that TRPS1 levels in RD cells, the bona fide model cell line for embryonal rhabdomyosarcoma, are regulated by miR-1 and that TRPS1 and MYOD1 share common genomic binding sites. The myogenin (MYOG) promoter is one of the critical targets of TRPS1 and MYOD1; we demonstrate that TRPS1 restricts MYOG expression and thereby inhibits terminal myogenic differentiation. Therefore, reduction of TRPS1 levels in embryonal rhabdomyosarcoma might be a therapeutic approach to drive embryonal rhabdomyosarcoma cells into myogenic differentiation, thereby generating postmitotic myotubes.
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