نتایج جستجو برای: myofibrillar myopathy

تعداد نتایج: 14255  

Journal: :Neuromuscular disorders : NMD 2015
Conrad C Weihl Robert H Baloh Youjin Lee Tsui-Fen Chou Sara K Pittman Glenn Lopate Peggy Allred Jennifer Jockel-Balsarotti Alan Pestronk Matthew B Harms

Sporadic inclusion body myositis (sIBM) has clinical, pathologic and pathomechanistic overlap with some inherited muscle and neurodegenerative disorders. In this study, DNA from 79 patients with sIBM was collected and the sequencing of 38 genes associated with hereditary inclusion body myopathy (IBM), myofibrillar myopathy, Emery-Dreifuss muscular dystrophy, distal myopathy, amyotrophic lateral...

2017
Andreas Unger Lisa Beckendorf Pierre Böhme Rudolf Kley Marion von Frieling-Salewsky Hanns Lochmüller Rolf Schröder Dieter O. Fürst Matthias Vorgerd Wolfgang A. Linke

Myopathies encompass a wide variety of acquired and hereditary disorders. The pathomechanisms include structural and functional changes affecting, e.g., myofiber metabolism and contractile properties. In this study, we observed increased passive tension (PT) of skinned myofibers from patients with myofibrillar myopathy (MFM) caused by FLNC mutations (MFM-filaminopathy) and limb-girdle muscular ...

Journal: :Brain : a journal of neurology 2007
R Griggs A Vihola P Hackman K Talvinen H Haravuori G Faulkner B Eymard I Richard D Selcen A Engel O Carpen B Udd

Distal myopathies have been associated with mutations in titin, dysferlin, GNE, desmin and myosin. Of these, only titin mutations were previously known to cause dominant late-onset distal myopathy. Recent findings, however, have indicated that patients affected with myofibrillar myopathy have a more distal than proximal muscle phenotype and a proportion of these may have mutations in myotilin, ...

Journal: :The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques 2014
Gerald Pfeffer Jeffrey T Joseph A Micheil Innes J Bevan Frizzell Ian J Wilson A Keith W Brownell Patrick F Chinnery

Hereditary myopathy with early respiratory failure (HMERF, OMIM 603689) is an autosomal dominant myopathy, often characterised by respiratory muscle weakness early in the disease course. This condition shares many similarities with myofibrillar myopathy (MFM) and it has been suggested that HMERF be considered within this disease category1,2. It is caused by mutations in TTN, which encodes the g...

Journal: :Human molecular genetics 2014
Andrea A Domenighetti Pao-Hsien Chu Tongbin Wu Farah Sheikh David S Gokhin Ling T Guo Ziyou Cui Angela K Peter Danos C Christodoulou Michael G Parfenov Joshua M Gorham Daniel Y Li Indroneal Banerjee Xianyin Lai Frank A Witzmann Christine E Seidman Jonathan G Seidman Aldrin V Gomes G Diane Shelton Richard L Lieber Ju Chen

Recent human genetic studies have provided evidences that sporadic or inherited missense mutations in four-and-a-half LIM domain protein 1 (FHL1), resulting in alterations in FHL1 protein expression, are associated with rare congenital myopathies, including reducing body myopathy and Emery-Dreifuss muscular dystrophy. However, it remains to be clarified whether mutations in FHL1 cause skeletal ...

Journal: :Journal of neuropathology and experimental neurology 2009
Montse Olivé Anna Janué Dolores Moreno Josep Gámez Benjamín Torrejón-Escribano Isidre Ferrer

Protein aggregate myopathies, including myofibrillar myopathies and sporadic inclusion body myositis (sIBM), are characterized by abnormal protein aggregates composed of various muscular and ectopic proteins. Previous studies have shown the crucial role ofdysregulated transcription factors such as neuron-restrictive silencerfactor in the expression of aberrant proteins in myotilinopathies. Here...

Journal: :Cell biology international 2015
Lilli Winter Wolfgang H Goldmann

Myofibrillar myopathies (MFMs) are a group of sporadic and hereditary skeletal muscle diseases, which lead to severe physical disability and premature death. Most MFMs are caused by mutations in genes encoding desmin, plectin, VCP, filamin C, BAG3, FHL-1, αB-crystallin, DNAJB6, myotilin, and ZASP. Biomechanical studies on primary human myoblasts carrying desmin and plectin mutations showed incr...

2015
Eva Cabet Sabrina Batonnet-Pichon Florence Delort Blandine Gausserès Patrick Vicart Alain Lilienbaum Gerhard Wiche

Desminopathies, a subgroup of myofibrillar myopathies (MFMs), the progressive muscular diseases characterized by the accumulation of granulofilamentous desmin-positive aggregates, result from mutations in the desmin gene (DES), encoding a muscle-specific intermediate filament. Desminopathies often lead to severe disability and premature death from cardiac and/or respiratory failure; no specific...

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