ArticleFrontiers in cell and developmental biology2022
Long-term culture of patient-derived cardiac organoids recapitulated Duchenne muscular dystrophy cardiomyopathy and disease progression.
Article in Frontiers in cell and developmental biology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.
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Who cites it
34 citing papers in PubMed.
- Cardiac microphysiological systems in cardiovascular research: Construction paradigms, maturation trajectories, and translational frontiers.Bioengineering & translational medicine · 2026Review
- Review
- Conformable Microelectrode Arrays Integrated with a Scoop-Shaped Slide-Well for Dynamic Electrophysiological Profiling of Patient-Derived Cardiac Organoids.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Advanced in vitro cardiac models for drug evaluation: integration of organoids, engineered tissues, and microphysiological systems.Microsystems & nanoengineering · 2026Review
- Patient-Derived 3D Bioprinted Cardiac Organoid Constructs Reveal Key Pathological Features of Duchenne Muscular Dystrophy.Advanced healthcare materials · 2026Article
- A Hypoxia-reoxygenation Injury Model in Self-assembling Human Cardioids.Journal of visualized experiments : JoVE · 2026Article
- Current Trends in Duchenne Muscular Dystrophy Research and Therapy: 3D Cardiac Modelling.Journal of cachexia, sarcopenia and muscle · 2026Review
- Human cardiac organoids: multidimensional integration and clinical translation potential.Frontiers in pharmacology · 2026Review
- Human Cardiac Organoids: Advances and Prospects from Construction to Preclinical Drug Evaluation.Cells · 2025Review
- [Construction of cardiac organoids derived from human induced pluripotent stem cells for cardiac disease modeling and drug evaluation].Nan fang yi ke da xue xue bao = Journal of Southern Medical University · 2025Article
- Review
- Cardiac Tissue Engineering for Translational Cardiology: From In Vitro Models to Regenerative Therapies.Bioengineering (Basel, Switzerland) · 2025Review
- Exploring organoid and assembloid technologies: a focus on retina and brain.Expert reviews in molecular medicine · 2025Review
- Revolutionizing cardiovascular research: Human organoids as a Beacon of hope for understanding and treating cardiovascular diseases.Materials today. Bio · 2025Review
- Application of human cardiac organoids in cardiovascular disease research.Frontiers in cell and developmental biology · 2025Review
- Human-based complexFrontiers in cell and developmental biology · 2025Review
- Investigating Inherited Heart Diseases Using Human Induced Pluripotent Stem Cell-Based Models.Life (Basel, Switzerland) · 2024Review
- Report and Abstracts of the 20th Meeting of IIM, the Interuniversity Institute of Myology: Assisi, October 12-15, 2023.European journal of translational myology · 2024Article
- Three-Dimensional iPSC-Based In Vitro Cardiac Models for Biomedical and Pharmaceutical Research Applications.International journal of molecular sciences · 2024Review
- The Pathogenic Mechanisms of and Novel Therapies for Lamin A/C-Related Dilated Cardiomyopathy Based on Patient-Specific Pluripotent Stem Cell Platforms and Animal Models.Pharmaceuticals (Basel, Switzerland) · 2024Review
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13 authors.
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Abstract
Duchenne Muscular Dystrophy (DMD) is an X-linked neuromuscular disease which to date is incurable. The major cause of death is dilated cardiomyopathy however, its pathogenesis is unclear as existing cellular and animal models do not fully recapitulate the human disease phenotypes. In this study, we generated cardiac organoids from patient-derived induced pluripotent stem cells (DMD-COs) and isogenic-corrected controls (DMD-Iso-COs) and studied if DMD-related cardiomyopathy and disease progression occur in the organoids upon long-term culture (up to 93 days). Histological analysis showed that DMD-COs lack initial proliferative capacity, displayed a progressive loss of sarcoglycan localization and high stress in endoplasmic reticulum. Additionally, cardiomyocyte deterioration, fibrosis and aberrant adipogenesis were observed in DMD-COs over time. RNA sequencing analysis confirmed a distinct transcriptomic profile in DMD-COs which was associated with functional enrichment in hypertrophy/dilated cardiomyopathy, arrhythmia, adipogenesis and fibrosis pathways. Moreover, five miRNAs were identified to be crucial in this dysregulated gene network. In conclusion, we generated patient-derived cardiac organoid model that displayed DMD-related cardiomyopathy and disease progression phenotypes in long-term culture. We envision the feasibility to develop a more complex, realistic and reliable
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