نتایج جستجو برای: col6a1

تعداد نتایج: 128  

2012
J. Kim C. Jimenez-Mallebrera A.R. Foley M. Fernandez-Fuente S.C. Brown S. Torelli L. Feng C.A. Sewry F. Muntoni

Mutations in COL6A1, COL6A2 and COL6A3 genes result in collagen VI myopathies: Ullrich congenital muscular dystrophy (UCMD), Bethlem myopathy (BM) and intermediate phenotypes. At present, none of the existing diagnostic techniques for evaluating collagen VI expression is quantitative, and the detection of subtle changes in collagen VI expression remains challenging. We investigated flow cytomet...

2017
Jérôme Bürgi Béatrice Kunz Laurence Abrami Julie Deuquet Alessandra Piersigilli Sabine Scholl-Bürgi Ekkehart Lausch Sheila Unger Andrea Superti-Furga Paolo Bonaldo F Gisou van der Goot

Loss-of-function mutations in capillary morphogenesis gene 2 (CMG2/ANTXR2), a transmembrane surface protein, cause hyaline fibromatosis syndrome (HFS), a severe genetic disorder that is characterized by large subcutaneous nodules, gingival hypertrophy and severe painful joint contracture. Here we show that CMG2 is an important regulator of collagen VI homoeostasis. CMG2 loss of function promote...

2015

Synonyms Spectrum of phenotypes: Mild: Bethlem myopathy/ benign congenital muscular dystrophy Intermediate: Limb-girdle muscular dystrophy; myosclerosis myopathy Severe: Ullrich myopathy/ congenital atonic sclerotic muscular dystrophy First described by Ullrich in 1930 and Bethlem in 1976 respectively [1]. Caused by mutations in any of the 3 genes which code for collagen type VI synthesis, COL6...

2017
Satoru Noguchi Megumu Ogawa May Christine Malicdan Ikuya Nonaka Ichizo Nishino

Congenital muscular dystrophies with collagen VI deficiency are inherited muscle disorders with a broad spectrum of clinical presentation and are caused by mutations in one of COL6A1-3 genes. Muscle pathology is characterized by fiber size variation and increased interstitial fibrosis and adipogenesis. In this study, we define critical events that contribute to muscle weakness and fibrosis in a...

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