ArticleeLife2015
In vivo dynamics of skeletal muscle Dystrophin in zebrafish embryos revealed by improved FRAP analysis.
Article in eLife, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.
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Who cites it
17 citing papers in PubMed, 31 citations in OpenAlex.
- Fishing for novel HDAC inhibitor compounds to treat Duchenne muscular dystrophy.Molecular therapy. Nucleic acids · 2025Article
- Spatiotemporal analysis of dystrophin expression during muscle repair.Skeletal muscle · 2025Article
- Gap-junction-mediated bioelectric signaling required for slow muscle development and function in zebrafish.Current biology : CB · 2024Article
- What's past is prologue: FRAP keeps delivering 50 years later.Biophysical journal · 2023Review
- Trichinella spiralis (Owen, 1835) Induces Increased Dystrophin Expression in Invaded Cross-striated Muscle.Acta parasitologica · 2023Article
- Dystrophin myonuclear domain restoration governs treatment efficacy in dystrophic muscle.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Evolution and developmental functions of the dystrophin-associated protein complex: beyond the idea of a muscle-specific cell adhesion complex.Frontiers in cell and developmental biology · 2023Review
- Nutraceutical Screening in a Zebrafish Model of Muscular Dystrophy: Gingerol as a Possible Food Aid.Nutrients · 2021Article
- Myogenin is an essential regulator of adult myofibre growth and muscle stem cell homeostasis.eLife · 2020Article
- Dystrophin As a Molecular Shock Absorber.ACS nano · 2018Article
- Fluorescence techniques in developmental biology.Journal of biosciences · 2018Review
- Dystrophin's central domain forms a complex filament that becomes disorganized by in-frame deletions.The Journal of biological chemistry · 2018Article
- Epigenetic Regulators Modulate Muscle Damage in Duchenne Muscular Dystrophy Model.PLoS currents · 2017Article
- The rise of photoresponsive protein technologies applicationsF1000Research · 2017Review
- Dystrophin contains multiple independent membrane-binding domains.Human molecular genetics · 2016Article
- Cellular dynamics of regeneration reveals role of two distinct Pax7 stem cell populations in larval zebrafish muscle repair.Disease models & mechanisms · 2016Article
- In vivo dynamics of skeletal muscle Dystrophin in zebrafish embryos revealed by improved FRAP analysis.eLife · 2015Article
Corrections and comments
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Authors and funding
8 authors at 5 institutions in 3 countries.
Funding
Abstract
Dystrophin forms an essential link between sarcolemma and cytoskeleton, perturbation of which causes muscular dystrophy. We analysed Dystrophin binding dynamics in vivo for the first time. Within maturing fibres of host zebrafish embryos, our analysis reveals a pool of diffusible Dystrophin and complexes bound at the fibre membrane. Combining modelling, an improved FRAP methodology and direct semi-quantitative analysis of bleaching suggests the existence of two membrane-bound Dystrophin populations with widely differing bound lifetimes: a stable, tightly bound pool, and a dynamic bound pool with high turnover rate that exchanges with the cytoplasmic pool. The three populations were found consistently in human and zebrafish Dystrophins overexpressed in wild-type or dmd(ta222a/ta222a) zebrafish embryos, which lack Dystrophin, and in Gt(dmd-Citrine)(ct90a) that express endogenously-driven tagged zebrafish Dystrophin. These results lead to a new model for Dystrophin membrane association in developing muscle, and highlight our methodology as a valuable strategy for in vivo analysis of complex protein dynamics.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.