نتایج جستجو برای: d4z4

تعداد نتایج: 154  

2011
Stefania Cheli Stephanie François Beatrice Bodega Francesco Ferrari Elena Tenedini Enrica Roncaglia Sergio Ferrari Enrico Ginelli Raffaella Meneveri

BACKGROUND Determine global gene dysregulation affecting 4q-linked (FSHD-1) and non 4q-linked (FSHD-2) cells during early stages of myogenic differentiation. This approach has been never applied to FSHD pathogenesis. METHODOLOGY/PRINCIPAL FINDINGS By in vitro differentiation of FSHD-1 and FSHD-2 myoblasts and gene chip analysis we derived that gene expression profile is altered only in FSHD-1...

Journal: :Human molecular genetics 2003
Sara T Winokur Yi-Wen Chen Peter S Masny Jorge H Martin Jeffrey T Ehmsen Stephen J Tapscott Silvere M van der Maarel Yukiko Hayashi Kevin M Flanigan

The neuromuscular disorder facioscapulohumeral muscular dystrophy (FSHD) results from integral deletions of the subtelomeric repeat D4Z4 on chromosome 4q. A disruption of chromatin structure affecting gene expression is thought to underlie the pathophysiology. The global gene expression profiling of mature muscle tissue presented here provides the first insight into an FSHD-specific defect in m...

2012
Giorgio Tasca Mario Pescatori Mauro Monforte Massimiliano Mirabella Elisabetta Iannaccone Roberto Frusciante Tiziana Cubeddu Francesco Laschena Pierfrancesco Ottaviani Enzo Ricci

BACKGROUND Facioscapulohumeral muscular dystrophy (FSHD) is one of the most common muscular dystrophies and is characterized by a non-conventional genetic mechanism activated by pathogenic D4Z4 repeat contractions. By muscle Magnetic Resonance Imaging (MRI) we observed that T2-short tau inversion recovery (T2-STIR) sequences identify two different conditions in which each muscle can be found be...

2013
Edgardo Daniel Corona Daniela Jacquelin Laura Gatica Alberto Luis Rosa

DUX4 (Double Homeobox Protein 4) is a nuclear transcription factor encoded at each D4Z4 unit of a tandem-repeat array at human chromosome 4q35. DUX4 constitutes a major candidate pathogenic protein for facioscapulohumeral muscular dystrophy (FSHD), the third most common form of inherited myopathy. A low-level expression of DUX4 compromises cell differentiation in myoblasts and its overexpressio...

2012
Rasmus Ribel-Madsen Mario F. Fraga Stine Jacobsen Jette Bork-Jensen Ester Lara Vincenzo Calvanese Agustin F. Fernandez Martin Friedrichsen Birgitte F. Vind Kurt Højlund Henning Beck-Nielsen Manel Esteller Allan Vaag Pernille Poulsen

BACKGROUND Monozygotic twins discordant for type 2 diabetes constitute an ideal model to study environmental contributions to type 2 diabetic traits. We aimed to examine whether global DNA methylation differences exist in major glucose metabolic tissues from these twins. METHODOLOGY/PRINCIPAL FINDINGS Skeletal muscle (n = 11 pairs) and subcutaneous adipose tissue (n = 5 pairs) biopsies were c...

Journal: :Human molecular genetics 1996
S T Winokur U Bengtsson J C Vargas J J Wasmuth M R Altherr

Facioscapulohumeral muscular dystrophy (FSHD) is an autosomal dominant neuromuscular disease that has been linked to deletions within a tandem array of 3.2 kb repeats adjacent to the telomere of 4q. These repeats are also present in other locations in the human genome, including the short arms of all the acrocentric chromosomes. Here, we examine two models for the role of this repeat in FSHD. F...

Journal: :Free radical biology & medicine 2012
Ahmed Turki Maurice Hayot Gilles Carnac Fabien Pillard Emilie Passerieux Sébastien Bommart Eric Raynaud de Mauverger Gérald Hugon Joel Pincemail Sylvia Pietri Karen Lambert Alexandra Belayew Yegor Vassetzky Raul Juntas Morales Jacques Mercier Dalila Laoudj-Chenivesse

Facioscapulohumeral muscular dystrophy (FSHD), the most frequent muscular dystrophy, is an autosomal dominant disease. In most individuals with FSHD, symptoms are restricted to muscles of the face, arms, legs, and trunk. FSHD is genetically linked to contractions of the D4Z4 repeat array causing activation of several genes. One of these maps in the repeat itself and expresses the DUX4 (the doub...

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