نتایج جستجو برای: d4z4

تعداد نتایج: 154  

Journal: :Human molecular genetics 2015
Richard J L F Lemmers Jelle J Goeman Patrick J van der Vliet Merlijn P van Nieuwenhuizen Judit Balog Marianne Vos-Versteeg Pilar Camano Maria Antonia Ramos Arroyo Ivonne Jerico Mark T Rogers Daniel G Miller Meena Upadhyaya Jan J G M Verschuuren Adolfo Lopez de Munain Arregui Baziel G M van Engelen George W Padberg Sabrina Sacconi Rabi Tawil Stephen J Tapscott Bert Bakker Silvère M van der Maarel

Facioscapulohumeral muscular dystrophy (FSHD: MIM#158900) is a common myopathy with marked but largely unexplained clinical inter- and intra-familial variability. It is caused by contractions of the D4Z4 repeat array on chromosome 4 to 1-10 units (FSHD1), or by mutations in the D4Z4-binding chromatin modifier SMCHD1 (FSHD2). Both situations lead to a partial opening of the D4Z4 chromatin struct...

Journal: :Human mutation 2009
Jessica C de Greef Richard J L F Lemmers Baziel G M van Engelen Sabrina Sacconi Shannon L Venance Rune R Frants Rabi Tawil Silvère M van der Maarel

Facioscapulohumeral muscular dystrophy (FSHD), caused by partial deletion of the D4Z4 macrosatellite repeat on chromosome 4q, has a complex genetic and epigenetic etiology. To develop FSHD, D4Z4 contraction needs to occur on a specific genetic background. Only contractions associated with the 4qA161 haplotype cause FSHD. In addition, contraction of the D4Z4 repeat in FSHD patients is associated...

2009
Weihua Zeng Jessica C. de Greef Yen-Yun Chen Richard Chien Xiangduo Kong Heather C. Gregson Sara T. Winokur April Pyle Keith D. Robertson John A. Schmiesing Virginia E. Kimonis Judit Balog Rune R. Frants Alexander R. Ball Leslie F. Lock Peter J. Donovan Silvère M. van der Maarel Kyoko Yokomori

Facioscapulohumeral dystrophy (FSHD) is an autosomal dominant muscular dystrophy in which no mutation of pathogenic gene(s) has been identified. Instead, the disease is, in most cases, genetically linked to a contraction in the number of 3.3 kb D4Z4 repeats on chromosome 4q. How contraction of the 4qter D4Z4 repeats causes muscular dystrophy is not understood. In addition, a smaller group of FS...

2008
Koji Tsumagari Lixin Qi Kesmic Jackson Chunbo Shao Michelle Lacey Janet Sowden Rabi Tawil Vettaikorumakankav Vedanarayanan Melanie Ehrlich

DNA methylation and chromatin DNaseI sensitivity were analyzed in and adjacent to D4Z4 repeat arrays, which consist of 1 to approximately 100 tandem 3.3-kb units at subtelomeric 4q and 10q. D4Z4 displayed hypomethylation in some cancers and hypermethylation in others relative to normal tissues. Surprisingly, in cancers with extensive D4Z4 methylation there was a barrier to hypermethylation spre...

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: :Cell 2002
Davide Gabellini Michael R. Green Rossella Tupler

Facioscapulohumeral muscular dystrophy (FSHD), a common myopathy, is an autosomal dominant disease of unknown molecular mechanism. Almost all FSHD patients carry deletions of an integral number of tandem 3.3 kilobase repeats, termed D4Z4, located on chromosome 4q35. Here, we find that in FSHD muscle, 4q35 genes located upstream of D4Z4 are inappropriately overexpressed. We show that an element ...

2014
Claudia Huichalaf Stefano Micheloni Giulia Ferri Roberta Caccia Davide Gabellini

Facioscapulohumeral muscular dystrophy (FSHD) is one of the most common inherited diseases of the skeletal muscle. It is characterized by asymmetric muscle weakness and variable penetrance. FSHD is linked to a reduction in copy number of the D4Z4 3.3 kb macrosatellite repeat, located in 4q35. This causes the epigenetic de-repression of FSHD candidate genes leading to disease. Nevertheless, the ...

2016
Sunny Das Brian P. Chadwick

We looked at a disease-associated macrosatellite array D4Z4 and focused on epigenetic factors influencing its chromatin state outside of the disease-context. We used the HCT116 cell line that contains the non-canonical polyadenylation (poly-A) signal required to stabilize somatic transcripts of the human double homeobox gene DUX4, encoded from D4Z4. In HCT116, D4Z4 is packaged into constitutive...

2012
Daphne S. Cabianca Valentina Casa Beatrice Bodega Alexandros Xynos Enrico Ginelli Yujiro Tanaka Davide Gabellini

Repetitive sequences account for more than 50% of the human genome. Facioscapulohumeral muscular dystrophy (FSHD) is an autosomal-dominant disease associated with reduction in the copy number of the D4Z4 repeat mapping to 4q35. By an unknown mechanism, D4Z4 deletion causes an epigenetic switch leading to de-repression of 4q35 genes. Here we show that the Polycomb group of epigenetic repressors ...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2006
Andrei Petrov Iryna Pirozhkova Gilles Carnac Dalila Laoudj Marc Lipinski Yegor S Vassetzky

Fascioscapulohumeral muscular dystrophy (FSHD) is an autosomal dominant neuromuscular disorder linked to partial deletion of integral numbers of a 3.3 kb polymorphic repeat, D4Z4, within the subtelomeric region of chromosome 4q. Although the relationship between deletions of D4Z4 and FSHD is well established, how this triggers the disease remains unclear. We have mapped the DNA loop domain cont...

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