نتایج جستجو برای: congenital myopathy

تعداد نتایج: 131548  

Journal: :Clinical genetics 2016
D Massalska J G Zimowski J Bijok A Kucińska-Chahwan A Łusakowska G Jakiel T Roszkowski

Congenital myopathies and muscular dystrophies constitute a genetically and phenotypically heterogeneous group of rare inherited diseases characterized by muscle weakness and atrophy, motor delay and respiratory insufficiency. To date, curative care is not available for these diseases, which may severely affect both life-span and quality of life. We discuss prenatal diagnosis and genetic counse...

2013
Asim Kumar Kundu

Freeman-Sheldon Syndrome (FSS), or distal arthrogryposis type 2A, is a rare congenital myopathy and dysplasia characterized by multiple contractures, abnormalities of the head and face, defective development of the hands and feet and skeletal malformations. The facial muscle contracture produces the typical “whistling face” appearance. Anesthetic issues include difficult intravenous access, dif...

2017
Silvio Alessandro Di Gioia Samantha Connors Norisada Matsunami Jessica Cannavino Matthew F. Rose Nicole M. Gilette Pietro Artoni Nara Lygia de Macena Sobreira Wai-Man Chan Bryn D. Webb Caroline D. Robson Long Cheng Carol Van Ryzin Andres Ramirez-Martinez Payam Mohassel Mark Leppert Mary Beth Scholand Christopher Grunseich Carlos R. Ferreira Tyler Hartman Ian M. Hayes Tim Morgan David M. Markie Michela Fagiolini Amy Swift Peter S. Chines Carlos E. Speck-Martins Francis S. Collins Ethylin Wang Jabs Carsten G. Bönnemann Eric N. Olson Caroline V. Andrews Brenda J. Barry David G. Hunter Sarah E. Mackinnon Sherin Shaaban Monica Erazo Tamiesha Frempong Ke Hao Thomas P. Naidich Janet C. Rucker Zhongyang Zhang Barbara B. Biesecker Lori L. Bonnycastle Carmen C. Brewer Brian P. Brooks John A. Butman Wade W. Chien Kathleen Farrell Edmond J. FitzGibbon Andrea L. Gropman Elizabeth B. Hutchinson Minal S. Jain Kelly A. King Tanya J. Lehky Janice Lee Denise K. Liberton Narisu Narisu Scott M. Paul Neda Sadeghi Joseph Snow Beth Solomon Angela Summers Camilo Toro Audrey Thurm Christopher K. Zalewski John C. Carey Stephen P. Robertson Irini Manoli Elizabeth C. Engle

Multinucleate cellular syncytial formation is a hallmark of skeletal muscle differentiation. Myomaker, encoded by Mymk (Tmem8c), is a well-conserved plasma membrane protein required for myoblast fusion to form multinucleated myotubes in mouse, chick, and zebrafish. Here, we report that autosomal recessive mutations in MYMK (OMIM 615345) cause Carey-Fineman-Ziter syndrome in humans (CFZS; OMIM 2...

2013
Joshua J.A. Lee Toshifumi Yokota

Antisense therapy is an approach to fighting diseases using short DNA-like molecules called antisense oligonucleotides. Recently, antisense therapy has emerged as an exciting and promising strategy for the treatment of various neurodegenerative and neuromuscular disorders. Previous and ongoing pre-clinical and clinical trials have provided encouraging early results. Spinal muscular atrophy (SMA...

Journal: :Science translational medicine 2014
Martin K Childers Romain Joubert Karine Poulard Christelle Moal Robert W Grange Jonathan A Doering Michael W Lawlor Branden E Rider Thibaud Jamet Nathalie Danièle Samia Martin Christel Rivière Thomas Soker Caroline Hammer Laetitia Van Wittenberghe Mandy Lockard Xuan Guan Melissa Goddard Erin Mitchell Jane Barber J Koudy Williams David L Mack Mark E Furth Alban Vignaud Carole Masurier Fulvio Mavilio Philippe Moullier Alan H Beggs Anna Buj-Bello

Loss-of-function mutations in the myotubularin gene (MTM1) cause X-linked myotubular myopathy (XLMTM), a fatal, congenital pediatric disease that affects the entire skeletal musculature. Systemic administration of a single dose of a recombinant serotype 8 adeno-associated virus (AAV8) vector expressing murine myotubularin to Mtm1-deficient knockout mice at the onset or at late stages of the dis...

2013
Young-Mi Han Kyoung-Ah Kwon Yun-Jin Lee Sang-Ook Nam Kyung-Hee Park Shin-Yun Byun Gu-Hwan Kim Han-Wook Yoo

X-linked recessive myotubular myopathy (XLMTM) is a severe congenital muscle disorder caused by mutations in the MTM1 gene and characterized by severe hypotonia and generalized muscle weakness in affected males. It is generally a fatal disorder during the neonatal period and early infancy. The diagnosis is based on typical histopathological findings on muscle biopsy, combined with suggestive cl...

بینش, فریبا, فلاح, راضیه, مرتضوی زاده, محمد رضا,

Mitochondrial myopathy, lactic acidosis, and siderobastic anemia (MLA SA) syndrome is one of the newly reported mitochondrial diseases, seven cases of which have been reported. We report a child with inflammatory myopathy, sideroblastic anemia and lactic acidosis .The patient is a 8.5 year old boy with normal cognitive function suffering from chronic progressive weakness in lower extremities, ...

Journal: :Journal of Korean Medical Science 1989
J. G. Chi H. S. Koo J. K. Roh

All the diagnostic muscle biopsy cases were collected from the file of Department of Pathology, Seoul National University Hospital during June 1976 to December 1978. Slides were reviewed and correlated with clinical informations. Two hundred seventy four cases showed pathological changes, which were classified into six large groups (Table 1). Neurogenic atrophy was most common, 97 cases (35%), ...

Journal: :Muscles, ligaments and tendons journal 2013
Francesca Tagliavini Francesca Sardone Stefano Squarzoni Nadir Mario Maraldi Luciano Merlini Cesare Faldini Patrizia Sabatelli

Collagen VI is an extracellular matrix protein expressed in several tissues including skeletal muscle. Mutations in COL6A genes cause Bethlem Myopathy (BM), Ullrich Congenital Muscular Dystrophy (UCMD) and Myosclerosis Myopathy (MM). Collagen VI deficiency causes increased opening of the mitochondrial permeability transition pore (mPTP), leading to ultrastructural and functional alterations of ...

Journal: :Journal of medical genetics 1977
U Ahmad E R Fisher T S Danowski S Nolan T Stephan

Abnormal endocrine indices and myopathy have been variably present in two brothers with Bloom's syndrome (congenital teleangiectatic erythema, hypersensitivity to light, and growth retardation). These consisted of: (1) growth retardation with height and weight below the third centiles; in the younger one at age 14, hypoglycaemia failed to elicit a rise in growth hormone but did so in the older ...

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