نتایج جستجو برای: dystrophin

تعداد نتایج: 3503  

Journal: :FEBS letters 1998
R H Crosbie H Yamada D P Venzke M P Lisanti K P Campbell

The dystrophin-glycoprotein complex is a multi-subunit protein complex that spans the muscle plasma membrane (sarcolemma) and forms a link between the intracellular cytoskeleton and the extracellular matrix. Caveolin-3, the muscle specific form of caveolin, is also a major structural and regulatory integral membrane protein found at the sarcolemma. Oligomers of caveolin-3 form the structural fr...

2012
Sue Fletcher Carl F. Adkin Penny Meloni Brenda Wong Francesco Muntoni Ryszard Kole Clayton Fragall Kane Greer Russell Johnsen Steve D. Wilton

Protein-truncating mutations in the dystrophin gene lead to the progressive muscle wasting disorder Duchenne muscular dystrophy, whereas in-frame deletions typically manifest as the milder allelic condition, Becker muscular dystrophy. Antisense oligomer-induced exon skipping can modify dystrophin gene expression so that a disease-associated dystrophin pre-mRNA is processed into a Becker muscula...

Journal: :The Journal of Cell Biology 1992
D Houzelstein G E Lyons J Chamberlain M E Buckingham

The spatial and temporal expression of the dystrophin gene has been examined during mouse embryogenesis, using in situ hybridization on tissue sections with a probe from the 5' end of the dystrophin coding sequence. In striated muscle, dystrophin transcripts are detectable from about 9 d in the heart and slightly later in skeletal muscle. However, there is an important difference between the tw...

2009
Maria Kinali Virginia Arechavala-Gomeza Lucy Feng Sebahattin Cirak David Hunt Carl Adkin Michela Guglieri Emma Ashton Stephen Abbs Petros Nihoyannopoulos Maria Elena Garralda Mary Rutherford Caroline Mcculley Linda Popplewell Ian R Graham George Dickson Matthew JA Wood Dominic J Wells Steve D Wilton Ryszard Kole Volker Straub Kate Bushby Caroline Sewry Jennifer E Morgan Francesco Muntoni

BACKGROUND Mutations that disrupt the open reading frame and prevent full translation of DMD, the gene that encodes dystrophin, underlie the fatal X-linked disease Duchenne muscular dystrophy. Oligonucleotides targeted to splicing elements (splice switching oligonucleotides) in DMD pre-mRNA can lead to exon skipping, restoration of the open reading frame, and the production of functional dystro...

Journal: :The Biochemical journal 1993
M Luise C Presotto L Senter R Betto S Ceoldo S Furlan S Salvatori R A Sabbadini G Salviati

Dystrophin, the protein coded by the gene missing in Duchenne muscular dystrophy, is assumed to be a component of the membrane cytoskeleton of skeletal muscle. Like other cytoskeletal proteins in different cell types, dystrophin bound to sarcolemma membranes was found to be phosphorylated by endogenous protein kinases. The phosphorylation of dystrophin was activated by cyclic AMP, cyclic GMP, c...

2018
Valentina Sardone Matthew Ellis Silvia Torelli Lucy Feng Darren Chambers Deborah Eastwood Caroline Sewry Rahul Phadke Jennifer E Morgan Francesco Muntoni

Clinical trials using strategies aimed at inducing dystrophin expression in Duchenne muscular dystrophy (DMD) are underway or at advanced planning stage, including splice switching antisense oligonucleotides (AON), drugs to induce read-through of nonsense mutations and viral mediated gene therapy. In all these strategies, different dystrophin proteins, often internally deleted, are produced, si...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2005
Qi Long Lu Adam Rabinowitz Yun Chao Chen Toshifumi Yokota HaiFang Yin Julia Alter Atif Jadoon George Bou-Gharios Terence Partridge

Antisense oligonucleotide-mediated alternative splicing has great potential for treatment of Duchenne muscular dystrophy (DMD) caused by mutations within nonessential regions of the dystrophin gene. We have recently shown in the dystrophic mdx mouse that exon 23, bearing a nonsense mutation, can be skipped after intramuscular injection of a specific 2'-O-methyl phosphorothioate antisense oligor...

Journal: :The Journal of Cell Biology 1995
J T Vilquin E Wagner I Kinoshita R Roy J P Tremblay

Myoblast transplantation has been considered a potential treatment for some muscular disorders. It has proven very successful, however, only in immunodeficient or immunosuppressed mice. In this study, myoblasts from C57BL10J +/+ mice were transplanted, with no immunosuppressive treatment, in the tibialis anterior of fully histocompatible but dystrophin-deficient C57BL10J mdx/mdx mice. One to 9 ...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2015
Dana M Talsness Joseph J Belanto James M Ervasti

The 427-kDa protein dystrophin is expressed in striated muscle where it physically links the interior of muscle fibers to the extracellular matrix. A range of mutations in the DMD gene encoding dystrophin lead to a severe muscular dystrophy known as Duchenne (DMD) or a typically milder form known as Becker (BMD). Patients with nonsense mutations in dystrophin are specifically targeted by stop c...

Journal: :Brain : a journal of neurology 2011
Karen Anthony Sebahattin Cirak Silvia Torelli Giorgio Tasca Lucy Feng Virginia Arechavala-Gomeza Annarita Armaroli Michela Guglieri Chiara S Straathof Jan J Verschuuren Annemieke Aartsma-Rus Paula Helderman-van den Enden Katherine Bushby Volker Straub Caroline Sewry Alessandra Ferlini Enzo Ricci Jennifer E Morgan Francesco Muntoni

Duchenne muscular dystrophy is caused by mutations in the DMD gene that disrupt the open reading frame and prevent the full translation of its protein product, dystrophin. Restoration of the open reading frame and dystrophin production can be achieved by exon skipping using antisense oligonucleotides targeted to splicing elements. This approach aims to transform the Duchenne muscular dystrophy ...

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