نتایج جستجو برای: lysosomal myopathy

تعداد نتایج: 27249  

2015
Lucia Marseglia Gabriella D’Angelo Sara Manti Vincenzo Salpietro Teresa Arrigo Vittorio Cavallari Eloisa Gitto

BACKGROUND Nemaline myopathy is a rare, non progressive congenital skeletal muscle disorder defined by the presence of inclusions known as nemaline rods in muscle fibers. Several clinical subtypes have been described, according to degree of muscle weakness, severity and age at onset. The course of nemaline myopathy is very slowly progressive, and death is usually due to respiratory failure. Car...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2002
Anna Wredenberg Rolf Wibom Hans Wilhelmsson Caroline Graff Heidi H Wiener Steven J Burden Anders Oldfors Håkan Westerblad Nils-Göran Larsson

We have generated an animal model for mitochondrial myopathy by disrupting the gene for mitochondrial transcription factor A (Tfam) in skeletal muscle of the mouse. The knockout animals developed a myopathy with ragged-red muscle fibers, accumulation of abnormally appearing mitochondria, and progressively deteriorating respiratory chain function in skeletal muscle. Enzyme histochemistry, electr...

2016
Metha Apiwattanakul Margherita Milone Sean J. Pittock Thomas J. Kryzer James P. Fryer Orna O'toole Andrew Mckeon Vanda A. Lennon

INTRODUCTION Paraneoplastic autoantibody screening of 150,000 patient sera by tissue-based immunofluorescence incidentally revealed 170 with unsuspected signal recognition particle (SRP) immunoglobulin G (IgG), which is a recognized biomarker of autoimmune myopathy. Of the 77 patients with available information, 54 had myopathy. We describe the clinical/laboratory associations. METHODS Distin...

2016
Josef Finsterer Claudia Stöllberger

OBJECTIVES Arrhythmogenic right ventricular dysplasia (ARVD) is a rare, genetic disorder predominantly affecting the right ventricle. There is increasing evidence that in some cases, ARVD is due to mutations in genes, which have also been implicated in primary myopathies. This review gives an overview about myopathy-associated ARVD and how these patients can be managed. METHODS A literature r...

Journal: :Annals of child neurology 2022

This corrects the article "Clinical Characteristics and Neurologic Outcomes of X-Linked Myotubular Myopathy" on page 127.

Journal: :Postgraduate medical journal 1987
R A Sheehan-Dare A V Simmons

A 57 year old female presented with an amyloid myopathy in association with lambda light chain myeloma. Treatment with melphalan and prednisolone resulted in remission of both myeloma and myopathy.

Journal: :Neurology 2004
Nortina Shahrizaila James Lowe Adrian Wills

The authors describe familial tubular aggregate myopathy associated with abnormal pupils. Four family members from two generations had myopathy and pupillary abnormalities. The myopathologic findings consisted of tubular aggregates in many fibers but predominantly type I fibers.

Journal: :British medical journal 1974
L R Baker P Ackrill W R Cattell T C Stamp L Watson

In a patient receiving regular dialysis prolonged hypophosphataemia due to aluminium hydroxide therapy resulted in osteomalacia and severe proximal myopathy. Both osteomalacia and myopathy responded to correction of hypophosphataemia without vitamin D therapy.

Journal: :Annals of neurology 2016
Hyung Jun Park Young Bin Hong Young-Chul Choi Jinho Lee Eun Ja Kim Ji-Su Lee Won Min Mo Soo Mi Ki Hyo In Kim Hye Jin Kim Young Se Hyun Hyun Dae Hong Kisoo Nam Sung Chul Jung Sang-Beom Kim Se Hoon Kim Deok-Ho Kim Ki-Wook Oh Seung Hyun Kim Jeong Hyun Yoo Ji Eun Lee Ki Wha Chung Byung-Ok Choi

OBJECTIVE Distal myopathy is a heterogeneous group of muscle diseases characterized by predominant distal muscle weakness. A study was done to identify the underlying cause of autosomal recessive adolescent onset distal myopathy. METHODS Four patients from 2 unrelated Korean families were evaluated. To isolate the genetic cause, exome sequencing was performed. In vitro and in vivo assays usin...

2003
Henna Haravuori Hannu Somer

Academic Dissertation To be publicly discussed with permission of the Medical Faculty of the University of Helsinki, in the small lecture hall of the Haartman Institute, This thesis is based on the following original articles, which are referred to in the text by their Roman numerals. In addition, some unpublished data are presented. Secondary calpain3 deficiency in 2q-linked muscular dystrophy...

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