نتایج جستجو برای: dystrophin deletions

تعداد نتایج: 22547  

Journal: :Journal of molecular biology 2012
Ava Yun Lin Ewa Prochniewicz Davin M Henderson Bin Li James M Ervasti David D Thomas

We have used time-resolved phosphorescence anisotropy (TPA) of actin to evaluate domains of dystrophin and utrophin, with implications for gene therapy in muscular dystrophy. Dystrophin and its homolog utrophin bind to cytoskeletal actin to form mechanical linkages that prevent muscular damage. Because these proteins are too large for most gene therapy vectors, much effort is currently devoted ...

Journal: :The Journal of molecular diagnostics : JMD 2011
Lisa Kalman Jay Leonard Norman Gerry Jack Tarleton Christina Bridges Julie M Gastier-Foster Robert E Pyatt Eileen Stonerock Monique A Johnson C Sue Richards Iris Schrijver Tianhui Ma Vanessa Rangel Miller Yetsa Adadevoh Pat Furlong Christine Beiswanger Lorraine Toji

Duchenne and Becker muscular dystrophies (DMD/BMD) are allelic X-linked recessive disorders that affect approximately 1 in 3500 and 1 in 20,000 male individuals, respectively. Approximately 65% of patients with DMD have deletions, 7% to 10% have duplications, and 25% to 30% have point mutations in one or more of the 79 exons of the dystrophin gene. Most clinical genetics laboratories test for d...

2017
D. Jake VanBelzen Alock S. Malik Paula S. Henthorn Joe N. Kornegay Hansell H. Stedman

Duchenne muscular dystrophy (DMD) is a lethal, X-linked, muscle-wasting disorder caused by mutations in the large, 2.4-Mb dystrophin gene. The majority of DMD-causing mutations are sporadic, multi-exon, frameshifting deletions, with the potential for variable immunological tolerance to the dystrophin protein from patient to patient. While systemic gene therapy holds promise in the treatment of ...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2012
Yoshitsugu Aoki Toshifumi Yokota Tetsuya Nagata Akinori Nakamura Jun Tanihata Takashi Saito Stephanie M R Duguez Kanneboyina Nagaraju Eric P Hoffman Terence Partridge Shin'ichi Takeda

Duchenne muscular dystrophy (DMD), the commonest form of muscular dystrophy, is caused by lack of dystrophin. One of the most promising therapeutic approaches is antisense-mediated elimination of frame-disrupting mutations by exon skipping. However, this approach faces two major hurdles: limited applicability of each individual target exon and uncertain function and stability of each resulting ...

Journal: :Journal of medical genetics 1992
L Vitiello M L Mostacciuolo S Oliviero F Schiavon L Nicoletti C Angelini G A Danieli

Mutations in the muscle promoter region and exonic deletions were screened in a series of 115 unrelated DMD and BMD patients from north-east Italy. No gross mutations of the promoter region were found. In three cases in which dystrophin of normal size was expressed at low levels, the analysis of DNA sequences of the promoter region failed to detect abnormalities. The majority of deletions in co...

Journal: :Neurologia 2017
I Vieitez P Gallano L González-Quereda S Borrego I Marcos J M Millán T Jairo C Prior J Molano M J Trujillo-Tiebas J Gallego-Merlo M García-Barcina M Fenollar C Navarro

INTRODUCTION Duchenne muscular dystrophy (DMD) is a severe X-linked recessive neuromuscular disease that affects one in 3500 live-born males. The total absence of dystrophin observed in DMD patients is generally caused by mutations that disrupt the reading frame of the DMD gene, and about 80% of cases harbour deletions or duplications of one or more exons. METHODS We reviewed 284 cases of mal...

2012
Corinne Betts Amer F Saleh Andrey A Arzumanov Suzan M Hammond Caroline Godfrey Thibault Coursindel Michael J Gait Matthew JA Wood

Antisense oligonucleotides (AOs) are currently the most promising therapeutic intervention for Duchenne muscular dystrophy (DMD). AOs modulate dystrophin pre-mRNA splicing, thereby specifically restoring the dystrophin reading frame and generating a truncated but semifunctional dystrophin protein. Challenges in the development of this approach are the relatively poor systemic AO delivery and in...

2008
Thanyachai Sura Jakris Eu-ahsunthornwattana Sarinee Pingsuthiwong Manisa Busabaratana

BACKGROUND Duchenne muscular dystrophy (DMD), a lethal X-linked disease affecting 1 in 3500 male births, and its more benign variant, Becker muscular dystrophy (BMD), are caused by mutations in the dystrophin gene. Because of its large size, analysing the whole gene is impractical. Methods have been developed to detect the commonest mutations i.e. the deletions of the exons. Although these test...

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