نتایج جستجو برای: dystrophin gene

تعداد نتایج: 1142885  

Journal: :Cardiovascular research 2010
Ralf Bauer Hugo A Katus Oliver J Müller

The dystrophin gene encodes an essential component of the transmembrane dystrophin–glycoprotein complex (DGC), which plays a critical role in maintaining membrane stability in cardiac and skeletal muscles. Defects in dystrophin destabilize the entire complex, which results in an abnormal susceptibility to sarcolemmal injury in response to contractile stress. Mutations that cause slight sequence...

Journal: :The Journal of clinical investigation 1999
E R Barton-Davis L Cordier D I Shoturma S E Leland H L Sweeney

Duchenne muscular dystrophy (DMD) is caused by mutations in the dystrophin gene, leading to the absence of the dystrophin protein in striated muscle. A significant number of these mutations are premature stop codons. On the basis of the observation that aminoglycoside treatment can suppress stop codons in cultured cells, we tested the effect of gentamicin on cultured muscle cells from the mdx m...

2014
Marly C Silva Carlos H Rassi Zilda M Meira Juliana G Giannetti Mariz Vainzof Mayana Zatz Roberto Kalil Carlos E Rochitte

Background Duchenne (DMD) and Becker (BMD) muscular dystrophies (MD) are allelic X-linked recessive disorders, caused by mutation of the dystrophin gene located at locus Xp21 that consists of 79 exons, characterized by progressive skeletal muscle degeneration and replacement by fibro fatty tissue. Dystrophin is a sarcolemal protein that links the cytoskeleton to the basal lamina and is essentia...

2017
Yukiya Sako Kensuke Ninomiya Yukiko Okuno Masayasu Toyomoto Atsushi Nishida Yuka Koike Kenji Ohe Isao Kii Suguru Yoshida Naohiro Hashimoto Takamitsu Hosoya Masafumi Matsuo Masatoshi Hagiwara

Duchenne muscular dystrophy (DMD) is a fatal progressive muscle-wasting disease. Various attempts are underway to convert severe DMD to a milder phenotype by modulating the splicing of the dystrophin gene and restoring its expression. In our previous study, we reported TG003, an inhibitor of CDC2-like kinase 1 (CLK1), as a splice-modifying compound for exon-skipping therapy; however, its metabo...

Journal: :EMBO reports 2004
Kristen J Nowak Kay E Davies

Duchenne muscular dystrophy (DMD) is caused by mutations in the gene that encodes the 427-kDa cytoskeletal protein dystrophin. Increased knowledge of the function of dystrophin and its role in muscle has led to a greater understanding of the pathogenesis of DMD. This, together with advances in the genetic toolkit of the molecular biologist, are leading to many different approaches to treatment....

Journal: :Science 2004
Aurélie Goyenvalle Adeline Vulin Françoise Fougerousse France Leturcq Jean-Claude Kaplan Luis Garcia Olivier Danos

Most mutations in the dystrophin gene create a frameshift or a stop in the mRNA and are associated with severe Duchenne muscular dystrophy. Exon skipping that naturally occurs at low frequency sometimes eliminates the mutation and leads to the production of a rescued protein. We have achieved persistent exon skipping that removes the mutated exon on the dystrophin messenger mRNA of the mdx mous...

2013
Sofia Muses Jennifer E Morgan Dominic J. Wells

The Sleeping beauty (SB) system is a non-viral DNA based vector that has been used to stably integrate therapeutic genes into disease models. Here we report the SB system is capable of stably integrating the ΔR4R23/CTΔ micro-dystrophin gene into a conditionally immortal dystrophin deficient muscle cell-line, H2K SF1, a murine cell model for Duchenne muscular dystrophy. Genetically corrected H2K...

2011
Sofia Muses Jennifer E Morgan Dominic J. Wells

The Sleeping beauty (SB) system is a non-viral DNA based vector that has been used to stably integrate therapeutic genes into disease models. Here we report the SB system is capable of stably integrating the ΔR4-R23/CTΔ micro-dystrophin gene into a conditionally immortal dystrophin deficient muscle cell-line, H2K SF1, a murine cell model for Duchenne muscular dystrophy. Genetically corrected H2...

Journal: :Human molecular genetics 2013
Nikolai Klymiuk Andreas Blutke Alexander Graf Sabine Krause Katinka Burkhardt Annegret Wuensch Stefan Krebs Barbara Kessler Valeri Zakhartchenko Mayuko Kurome Elisabeth Kemter Hiroshi Nagashima Benedikt Schoser Nadja Herbach Helmut Blum Rüdiger Wanke Annemieke Aartsma-Rus Christian Thirion Hanns Lochmüller Maggie C Walter Eckhard Wolf

Duchenne muscular dystrophy (DMD) is caused by mutations in the X-linked dystrophin (DMD) gene. The absence of dystrophin protein leads to progressive muscle weakness and wasting, disability and death. To establish a tailored large animal model of DMD, we deleted DMD exon 52 in male pig cells by gene targeting and generated offspring by nuclear transfer. DMD pigs exhibit absence of dystrophin i...

Journal: :The New England journal of medicine 2007
Judith C van Deutekom Anneke A Janson Ieke B Ginjaar Wendy S Frankhuizen Annemieke Aartsma-Rus Mattie Bremmer-Bout Johan T den Dunnen Klaas Koop Anneke J van der Kooi Nathalie M Goemans Sjef J de Kimpe Peter F Ekhart Edna H Venneker Gerard J Platenburg Jan J Verschuuren Gert-Jan B van Ommen

BACKGROUND Duchenne's muscular dystrophy is associated with severe, progressive muscle weakness and typically leads to death between the ages of 20 and 35 years. By inducing specific exon skipping during messenger RNA (mRNA) splicing, antisense compounds were recently shown to correct the open reading frame of the DMD gene and thus to restore dystrophin expression in vitro and in animal models ...

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