ArticlePloS one2007
Utrophin up-regulation by an artificial transcription factor in transgenic mice.
Article in PloS one, 2007. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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Who cites it
22 citing papers in PubMed, 44 citations in OpenAlex.
- Therapeutic potential of highly functional codon-optimized microutrophin for muscle-specific expression.Scientific reports · 2022Article
- Therapeutic Strategies for Dystrophin Replacement in Duchenne Muscular Dystrophy.Frontiers in medicine · 2022Review
- PMO-based let-7c site blocking oligonucleotide (SBO) mediated utrophin upregulation in mdx mice, a therapeutic approach for Duchenne muscular dystrophy (DMD).Scientific reports · 2020Article
- Genome Editing-Mediated Utrophin Upregulation in Duchenne Muscular Dystrophy Stem Cells.Molecular therapy. Nucleic acids · 2020Article
- Utrophin up-regulation by artificial transcription factors induces muscle rescue and impacts the neuromuscular junction in mdx mice.Biochimica et biophysica acta. Molecular basis of disease · 2018Article
- ZFP226 is a novel artificial transcription factor for selective activation of tumor suppressor KIBRA.Scientific reports · 2018Article
- Artificial Zinc-Finger Transcription Factor of A20 Suppresses Restenosis in Sprague Dawley Rats after Carotid Injury via the PPARα Pathway.Molecular therapy. Nucleic acids · 2017Article
- RNA activation of haploinsufficient Foxg1 gene in murine neocortex.Scientific reports · 2016Article
- Upregulating endogenous genes by an RNA-programmable artificial transactivator.Nucleic acids research · 2015Article
- Prevention of exercised induced cardiomyopathy following Pip-PMO treatment in dystrophic mdx mice.Scientific reports · 2015Article
- High throughput screening in duchenne muscular dystrophy: from drug discovery to functional genomics.Biology · 2014Review
- Novel adeno-associated viral vector delivering the utrophin gene regulator jazz counteracts dystrophic pathology in mdx mice.Journal of cellular physiology · 2014Article
- Design and Development of Artificial Zinc Finger Transcription Factors and Zinc Finger Nucleases to the hTERT Locus.Molecular therapy. Nucleic acids · 2013Article
- UtroUp is a novel six zinc finger artificial transcription factor that recognises 18 base pairs of the utrophin promoter and efficiently drives utrophin upregulation.BMC molecular biology · 2013Article
- Transcription factor rational design improves directed differentiation of human mesenchymal stem cells into skeletal myocytes.Molecular therapy : the journal of the American Society of Gene Therapy · 2011Article
- Article
- Zinc-finger-based transcriptional repression of rhodopsin in a model of dominant retinitis pigmentosa.EMBO molecular medicine · 2011Article
- Novel compounds for the treatment of Duchenne muscular dystrophy: emerging therapeutic agents.The application of clinical genetics · 2011Article
- β1-syntrophin modulation by miR-222 in mdx mice.PloS one · 2010Article
- Zinc-finger nucleases: a powerful tool for genetic engineering of animals.Transgenic research · 2010Review
Corrections and comments
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Authors and funding
14 authors at 7 institutions in 2 countries.
Funding
Abstract
Duchenne Muscular Dystrophy (DMD) is a severe muscle degenerative disease, due to absence of dystrophin. There is currently no effective treatment for DMD. Our aim is to up-regulate the expression level of the dystrophin related gene utrophin in DMD, complementing in this way the lack of dystrophin functions. To this end we designed and engineered several synthetic zinc finger based transcription factors. In particular, we have previously shown that the artificial three zinc finger protein named Jazz, fused with the appropriate effector domain, is able to drive the transcription of a test gene from the utrophin promoter "A". Here we report on the characterization of Vp16-Jazz-transgenic mice that specifically over-express the utrophin gene at the muscular level. A Chromatin Immunoprecipitation assay (ChIP) demonstrated the effective access/binding of the Jazz protein to active chromatin in mouse muscle and Vp16-Jazz was shown to be able to up-regulate endogenous utrophin gene expression by immunohistochemistry, western blot analyses and real-time PCR. To our knowledge, this is the first example of a transgenic mouse expressing an artificial gene coding for a zinc finger based transcription factor. The achievement of Vp16-Jazz transgenic mice validates the strategy of transcriptional targeting of endogenous genes and could represent an exclusive animal model for use in drug discovery and therapeutics.
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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.