نتایج جستجو برای: fshd

تعداد نتایج: 347  

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...

Journal: :American journal of human genetics 2012
Isabella Scionti Francesca Greco Giulia Ricci Monica Govi Patricia Arashiro Liliana Vercelli Angela Berardinelli Corrado Angelini Giovanni Antonini Michelangelo Cao Antonio Di Muzio Maurizio Moggio Lucia Morandi Enzo Ricci Carmelo Rodolico Lucia Ruggiero Lucio Santoro Gabriele Siciliano Giuliano Tomelleri Carlo Pietro Trevisan Giuliana Galluzzi Woodring Wright Mayana Zatz Rossella Tupler

Facioscapulohumeral muscular dystrophy (FSHD) is a common hereditary myopathy causally linked to reduced numbers (≤8) of 3.3 kilobase D4Z4 tandem repeats at 4q35. However, because individuals carrying D4Z4-reduced alleles and no FSHD and patients with FSHD and no short allele have been observed, additional markers have been proposed to support an FSHD molecular diagnosis. In particular a reduct...

Journal: :genetics in the 3rd millennium 0
bita bozorgmehr mehdi vahid dastjerdi ariana kariminejad

facioscapulohumeral muscular dystrophy (fshd)is characterized by weakness of the facial, scapular muscles  and the dorsiflexors of the foot. severity in this disorder is highly variable. approximately 95% of individuals with fshd phenotype have type 1 fshd, with d4z4 allele of between one and ten repeat units and about 5% have type 2 fshd with mutations in the chromatin modifier smchd1gene whic...

Journal: :Human molecular genetics 2012
Takako Iida Jones Jennifer C J Chen Fedik Rahimov Sachiko Homma Patricia Arashiro Mary Lou Beermann Oliver D King Jeffrey B Miller Louis M Kunkel Charles P Emerson Kathryn R Wagner Peter L Jones

Facioscapulohumeral muscular dystrophy (FSHD), the most prevalent myopathy afflicting both children and adults, is predominantly associated with contractions in the 4q35-localized macrosatellite D4Z4 repeat array. Recent studies have proposed that FSHD pathology is caused by the misexpression of the DUX4 (double homeobox 4) gene resulting in production of a pathogenic protein, DUX4-FL, which ha...

2014
Ricci G Zatz M Tupler R

Facioscapulohumeral muscular dystrophy (FSHD) has been classified as an autosomal dominant myopathy, linked to rearrangements in an array of 3.3 kb tandemly repeated DNA elements (D4Z4) located at the 4q subtelomere (4q35). For the last 20 years, the diagnosis of FSHD has been confirmed in clinical practice by the detection of one D4Z4 allele with a reduced number (≤8) of repeats at 4q35. Altho...

Journal: :Human molecular genetics 2013
Hiroaki Mitsuhashi Satomi Mitsuhashi Taylor Lynn-Jones Genri Kawahara Louis M Kunkel

Facioscapulohumeral muscular dystrophy (FSHD) is a common form of muscular dystrophy characterized by an asymmetric progressive weakness and wasting of the facial, shoulder and upper arm muscles, frequently accompanied by hearing loss and retinal vasculopathy. FSHD is an autosomal dominant disease linked to chromosome 4q35, but the causative gene remains controversial. DUX4 is a leading candida...

2014
Stefano Pasotti Bruno Magnani Emanuela Longa Giuseppe Giovanetti Albino Rossi Angela Berardinelli Rossella Tupler Giuseppe D’Antona

BACKGROUND Muscle fatigue, weakness and atrophy are basilar clinical features that accompany facioscapulohumeral dystrophy (FSHD) the third most common muscular dystrophy.No therapy is available for FSHD. CASE PRESENTATION We describe the effects of 6mo exercise therapy and nutritional supplementation in a 43-year-old woman severely affected by FSHD. CONCLUSION A mixed exercise program comb...

Journal: :Journal of the Royal Society, Interface 2015
Christopher R S Banerji Paul Knopp Louise A Moyle Simone Severini Richard W Orrell Andrew E Teschendorff Peter S Zammit

Facioscapulohumeral muscular dystrophy (FSHD) is an incurable disease, characterized by skeletal muscle weakness and wasting. Genetically, FSHD is characterized by contraction or hypomethylation of repeat D4Z4 units on chromosome 4, which causes aberrant expression of the transcription factor DUX4 from the last repeat. Many genes have been implicated in FSHD pathophysiology, but an integrated m...

Journal: :American journal of human genetics 1995
M Zatz S K Marie M R Passos-Bueno M Vainzof S Campiotto A Cerqueira C Wijmenga G Padberg R Frants

A gene responsible for facioscapulohumeral muscular dystrophy (FSHD) has been localized at 4q35. Subsequently, it was found that probe p13E-11 detects a polymorphic EcoRI fragment, usually > 28 kb, in normal individuals, whereas in sporadic and familial FSHD cases, an EcoRI fragment, usually < 28 kb, was found. Although these findings have been amply confirmed, several aspects are as yet either...

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