نتایج جستجو برای: utrophin

تعداد نتایج: 527  

Journal: :Human molecular genetics 2011
Alastair Crisp Haifang Yin Aurelie Goyenvalle Corinne Betts Hong M Moulton Yiqi Seow Arran Babbs Thomas Merritt Amer F Saleh Michael J Gait Daniel J Stuckey Kieran Clarke Kay E Davies Matthew J A Wood

Duchenne muscular dystrophy (DMD) is an X-linked recessive disease caused, in most cases, by the complete absence of the 427 kDa cytoskeletal protein, dystrophin. There is no effective treatment, and affected individuals die from respiratory failure and cardiomyopathy by age 30. Here, we investigated whether cardiomyopathy could be prevented in animal models of DMD by increasing diaphragm utrop...

Journal: :Journal of Biological Chemistry 1995

Journal: :Proceedings of the National Academy of Sciences of the United States of America 1994
J M Tinsley D J Blake R A Zuellig K E Davies

Duchenne muscular dystrophy is a severe X chromosome-linked, muscle-wasting disease caused by lack of the protein dystrophin. The exact function of dystrophin remains to be determined. However, analysis of its interaction with a large oligomeric protein complex at the sarcolemma and the identification of a structurally related protein, utrophin, is leading to the characterization of candidate g...

2012
Paula V. Cabrera Mabel Pang Jamie L. Marshall Raymond Kung Stanley F. Nelson Stephanie H. Stalnaker Lance Wells Rachelle H. Crosbie-Watson Linda G. Baum

Duchenne muscular dystrophy is an X-linked disorder characterized by loss of dystrophin, a cytoskeletal protein that connects the actin cytoskeleton in skeletal muscle cells to extracellular matrix. Dystrophin binds to the cytoplasmic domain of the transmembrane glycoprotein β-dystroglycan (β-DG), which associates with cell surface α-dystroglycan (α-DG) that binds laminin in the extracellular m...

Journal: :Journal of molecular and cellular cardiology 1997
R D Bies M Maeda S L Roberds E Holder T Bohlmeyer J B Young K P Campbell

5'-mutations in the dystrophin gene can result in cardiomyopathy without clinically-apparent skeletal myopathy. The effect of dystrophin mutations on the assembly and stability of the dystrophin associated protein (DAP) complex in human heart are not fully understood. The molecular defect in the dystrophin complex was explored in a family with an X-linked pedigree and severe dilated cardiomyopa...

Journal: :Biochemistry 2015
Surinder M Singh Swati Bandi Krishna M G Mallela

Dystrophin and utrophin are two muscle proteins involved in Duchenne/Becker muscular dystrophy. Both proteins use tandem calponin-homology (CH) domains to bind to F-actin. We probed the role of N-terminal CH1 and C-terminal CH2 domains in the structure and function of dystrophin tandem CH domain and compared with our earlier results on utrophin to understand the unifying principles of how tande...

Journal: :American journal of physiology. Regulatory, integrative and comparative physiology 2013
Katrin Hollinger Delphine Gardan-Salmon Connie Santana Drance Rice Elizabeth Snella Joshua T Selsby

Duchenne muscular dystrophy is typically diagnosed in the preschool years because of locomotor defects, indicative of muscle damage. Thus, effective therapies must be able to rescue muscle from further decline. We have established that peroxisome proliferator-activated receptor gamma coactivator 1-alpha (Pgc-1α) gene transfer will prevent many aspects of dystrophic pathology, likely through upr...

Journal: :Histology and histopathology 2011
M Kálmán J Mahalek A Adorján I Adorján K Pócsai Z Bagyura S Sadeghian

Dystroglycan is a laminin receptor, which with dystrophins and other components forms the dystrophin-dystroglycan complex. It has an important role in the formation of gliovascular connections, cerebral vascularisation and blood-brain barrier. Dystroglycan consists of two sub-units, α and β. Previous studies demonstrated that the β-dystroglycan immunoreactivity of cerebral vessels temporarily d...

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