نتایج جستجو برای: spinal muscular atrophy sma

تعداد نتایج: 194162  

2013
Gerrit Haaker Albert Fujak

Spinal muscular atrophy (SMA) is a hereditary neuromuscular disease of lower motor neurons that is caused by a defective "survival motor neuron" (SMN) protein that is mainly associated with proximal progressive muscle weakness and atrophy. Although SMA involves a wide range of disease severity and a high mortality and morbidity rate, recent advances in multidisciplinary supportive care have enh...

Journal: :Journal of medical genetics 1991
Z Borochowitz B Glick S Blazer

Acute infantile spinal muscular atrophy (SMA type I, Werdnig-Hoffmann disease) has generally been accepted as an autosomal recessive disorder. However, several investigators have noted a slightly increased male to female ratio. We describe here a family with two affected male sibs who had a form of acute infantile SMA with congenital bone fractures, whose parents were first cousins. Pedigree an...

Journal: :Muscle & nerve 2012
Seward B Rutkove Matt C Gregas Basil T Darras

INTRODUCTION New approaches for assessing disease progression in spinal muscular atrophy (SMA) are needed. In this study, we evaluate whether electrical impedance myography (EIM) can detect disease progression in SMA compared with a group of healthy children of similar age. METHODS Twenty-eight children with SMA and 20 normal children underwent repeated EIM testing in four muscles at regular ...

Journal: :Human molecular genetics 2007
Chen-Hung Ting Chiao-Wei Lin Shin-Lan Wen Hsiu-Mei Hsieh-Li Hung Li

Proximal spinal muscular atrophy (SMA) is a motor neuron degeneration disorder for which there is currently no effective treatment. Here, we report three compounds (sodium vanadate, trichostatin A and aclarubicin) that effectively enhance SMN2 expression by inducing Stat5 activation in SMA-like mouse embryonic fibroblasts and human SMN2-transfected NSC34 cells. We found that Stat5 activation en...

Journal: :Muscle & nerve 2014
Stefan J Cano Anna Mayhew Allan M Glanzman Kristin J Krosschell Kathryn J Swoboda Marion Main Birgit F Steffensen Carole Bérard Françoise Girardot Christine A M Payan Eugenio Mercuri Elena Mazzone Bakri Elsheikh Julaine Florence Linda S Hynan Susan T Iannaccone Leslie L Nelson Shree Pandya Michael Rose Charles Scott Reza Sadjadi Mackensie A Yore Cynthia Joyce John T Kissel

INTRODUCTION Trial design for SMA depends on meaningful rating scales to assess outcomes. In this study Rasch methodology was applied to 9 motor scales in spinal muscular atrophy (SMA). METHODS Data from all 3 SMA types were provided by research groups for 9 commonly used scales. Rasch methodology assessed the ordering of response option thresholds, tests of fit, spread of item locations, res...

2006
A. MOOSA V. DUBOWITZ

Moosa, A., and Dubowitz, V. (1973). Archives of Disease in Childhood, 48, 386. Spinal muscular atrophy in childhood: two clues to clinical diagnosis. A coarse tremor was noted in 13 children suffering from the childhood form of spinal muscular atrophy. Tremor has not been seen in any other condition producing proximal muscle weakness in childhood, and its presence should therefore suggest the d...

Journal: :Journal of medical genetics 1992
R J Daniels G K Suthers K E Morrison N H Thomas M J Francis C G Mathew S Loughlin A Heiberg D Wood V Dubowitz

Spinal muscular atrophy (SMA) is a common cause of inherited morbidity and mortality in childhood. The wide range of phenotypes in SMA, uncertainty regarding its mode of inheritance, and the suggestion of linkage heterogeneity have complicated the genetic counselling of parents of affected children. The locus responsible for autosomal recessive SMA has been mapped to 5q11.2-q13.3. The most like...

2014
Jeremiah Hadwen Duncan MacKenzie Fahad Shamim Kevin Mongeon Martin Holcik Alex MacKenzie Faraz Farooq

BACKGROUND Spinal Muscular Atrophy (SMA) is one of the most common inherited causes of infant death and is caused by the loss of functional survival motor neuron (SMN) protein due to mutations or deletion in the SMN1 gene. One of the treatment strategies for SMA is to induce the expression of the protein from the homologous SMN2 gene, a rescuing paralog for SMA. METHODS AND RESULTS Here we de...

Journal: :Archives of neurology 2002
Susan T Iannaccone

BACKGROUND Spinal muscular atrophy (SMA) is a genetic disease of the anterior horn cell with a frequency of 8 per 100 000 live births and a high rate of mortality during infancy. The American Spinal Muscular Atrophy Randomized Trials (AmSMART) Group is an organization of 5 centers formed to perform clinical trials in children with SMA. OBJECTIVE To devise reliable methods to measure strength,...

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