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Myoblasts generated by lentiviral mediated MyoD transduction of myotonic dystrophy type 1 (DM1) fibroblasts can be used for assays of therapeutic molecules by Jan Larsen; Olof J Pettersson; Maria Jakobsen; Rune Thomsen; Christina B Pedersen; Jens M Hertz; Niels Gregersen; Thomas J Corydon; Thomas G Jensen is a Medicine article available to read on EtoBox.
What is Myoblasts generated by lentiviral mediated MyoD transduction of myotonic dystrophy type 1 (DM1) fibroblasts can be used for assays of therapeutic molecules about?
## Background Myotonic dystrophy type 1 (DM1) is the most common muscle dystrophy in adults. The disease is caused by a triplet expansion in the 3'end of the myotonic dystrophy protein kinase (__DMPK)__ gene. In order to develop a human cell model for investigation of possible effects of antisense and RNAi effector molecules we have used lentiviral mediated __myoD__-forced myogenesis of DM1 patient fibroblasts. ## Findings Transduced fibroblasts show a multinuclear phenotype and express the differentiation marker myogenin. Furthermore, fluorescence in situ hybridization (FISH) analysis revealed a statistical significant increase in the amount of nuclear foci in DM1 patient fibroblasts after myogenesis. Finally, no nuclear foci were found after treatment with oligonucleotides targeting the repeat expansions. ## Conclusions The abundance of nuclear foci in DM1 patient fibroblasts increase following myogenesis, as visualized by FISH analysis. Foci were eradicated after treatment with antisense oligonucleotides. Thus, we propose that the current cell model is suitable for testing of novel treatment modalities.
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- Author
- Jan Larsen; Olof J Pettersson; Maria Jakobsen; Rune Thomsen; Christina B Pedersen; Jens M Hertz; Niels Gregersen; Thomas J Corydon; Thomas G Jensen
- Publisher
- BioMed Central; Springer (Biomed Central Ltd.); London: Biomed Central, 2008-; Publons; Springer Science and Business Media LLC; Society for Mining, Metallurgy and Exploration Inc. (ISSN 1756-0500)
- Published
- 2011
- Language
- EN
- Field
- Medicine (Health Sciences)