نتایج جستجو برای: col6a1

تعداد نتایج: 128  

Journal: :iranian journal of child neurology 0
bita bozorgmehr kariminejad &najmabadi genetic center ariana kariminejad kariminejad & najmabadi genetic center, tehran, iran shahriar nafissi shariati hospital, tehran, iran bita jebelli pediatric neurologist, tehran, iran urtizberea andoni hospital marin, paris, france corine gartoux 5. upmc univ paris 06, ifr14, paris, f-75013, france 6. cnrs, umr7215, paris, f-75013, france 7. inserm, u974, paris, f-75013, france 8. institut de myologie, paris, f-75013, france

how to cite this article: bozorgmehr b, kariminejad a, nafissi sh, jebelli b, andoni u, gartioux c, ledeuil c, allamand y, richard p, kariminejad mh. ullrich congenital muscular dystrophy (ucmd):clinical and genetic correlations. iran j child neurol. 2013 summer; 7(3): 15-22.   obj ective: ullrich congenital muscular dystrophy (ucmd) corresponds to the severe end of the clinical spectrum of neu...

Journal: :Human molecular genetics 2007
Vanessa Besson Véronique Brault Arnaud Duchon Dieudonné Togbe Jean-Charles Bizot Valérie F J Quesniaux Bernard Ryffel Yann Hérault

Monosomy 21 is a rare human disease due to gene dosage errors disturbing a variety of physiological and morphological systems including brain, skeletal, immune and respiratory functions. Most of the human condition corresponds to partial or mosaic monosomy suggesting that Monosomy 21 may be lethal. In order to search for dosage-sensitive genes involved in the human pathology, we generated by ch...

Journal: :Arquivos de neuro-psiquiatria 2005
Umbertina Conti Reed Lucio Gobbo Ferreira Enna Cristina Liu Maria Bernadete Dutra Resende Mary Souza Carvalho Suely Kazue Marie Milberto Scaff

UNLABELLED Ullrich congenital muscular dystrophy (UCMD), due to mutations in the collagen VI genes, is an autosomal recessive form of CMD, commonly associated with distal joints hyperlaxity and severe course. A mild or moderate involvement can be occasionally observed. OBJECTIVE To evaluate the clinical picture of CMD patients with Ullrich phenotype who presented decreased or absent collagen ...

Journal: :Human molecular genetics 2014
Alessandra Zulian Erika Rizzo Marco Schiavone Elena Palma Francesca Tagliavini Bert Blaauw Luciano Merlini Nadir Mario Maraldi Patrizia Sabatelli Paola Braghetta Paolo Bonaldo Francesco Argenton Paolo Bernardi

Ullrich congenital muscular dystrophy (UCMD) and Bethlem myopathy (BM) are inherited muscle diseases due to mutations in the genes encoding the extracellular matrix protein collagen (Col) VI. Opening of the cyclosporin A-sensitive mitochondrial permeability transition pore (PTP) is a causative event in disease pathogenesis, and a potential target for therapy. Here, we have tested the effect of ...

Journal: :Cold Spring Harbor perspectives in biology 2013
Paolo Bernardi Paolo Bonaldo

Ullrich Congenital Muscular Dystrophy (UCMD), Bethlem Myopathy (BM), and Congenital Myosclerosis are diseases caused by mutations in the genes encoding the extracellular matrix protein collagen VI. A dystrophic mouse model, where collagen VI synthesis was prevented by targeted inactivation of the Col6a1 gene, allowed the investigation of pathogenesis, which revealed the existence of a Ca(2+)-me...

2005
In-Hyuk Chung Sook-Hwan Lee Kyo-Won Lee Sang-hee Park Kwang-Yul Cha Nam-Soon Kim Hyang-Sook Yoo Yong Sung Kim Suman Lee

Complete or partial triplication of human chromosome 21 results in Down syndrome (DS). To analyze differential gene expressions in amniotic fluid (AF) cells of DS, we used a DNA microarray system to analyze 102 genes, which included 24 genes on chromosome 21, 28 genes related to the function of brain and muscle, 36 genes related to apoptosis, 4 genes related to extracellular matrix, 8 genes rel...

Journal: :Journal of the Korean Neurological Association 2018

2010
W.R. Telfer A.S. Busta C.G. Bonnemann E.L. Feldman J.J. Dowling

Collagen VI is an integral part of the skeletal muscle extracellular matrix, providing mechanical stability and facilitating matrix-dependent cell signaling. Mutations in collagen VI result in either Ullrich congenital muscular dystrophy (UCMD) or Bethlem myopathy (BM), with UCMD being clinically more severe. Recent studies demonstrating increased apoptosis and abnormal mitochondrial function i...

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