نتایج جستجو برای: spinal muscular atrophy sma

تعداد نتایج: 194162  

2018
Aziza Alrafiah Evangelia Karyka Ian Coldicott Kayleigh Iremonger Katherin E. Lewis Ke Ning Mimoun Azzouz

Spinal muscular atrophy (SMA) is a devastating childhood motor neuron disease. SMA is caused by mutations in the survival motor neuron gene (SMN1), leading to reduced levels of SMN protein in the CNS. The actin-binding protein plastin 3 (PLS3) has been reported as a modifier for SMA, making it a potential therapeutic target. Here, we show reduced levels of PLS3 protein in the brain and spinal c...

2013
Borja Valencia John Robert Bach

INTRODUCTION SMA-1, also known as Werdnig-Hoffmann disease, is the most common neuromuscular disease of hypotonic newborns. Its incidence is over 1 per 8000 births and is a common cause of sudden infant death syndrome. It is characterized by the degeneration of the anterior horn cells of the spinal cord and of the motor nuclei in the lower brainstem (1). About 99% of patients have deletions in ...

2010
Gideon Dreyfuss Howard Hughes

Spinal muscular atrophy (SMA) is the most common motor neuron degenerative disease and is the principal genetic cause of infant mortality, affecting 1 in every 6000 newborns. The survival of motor neurons (SMN) gene has been implicated as the disease-causing gene in SMA, and it is deleted or mutated in over 98% of SMA patients. Our lab has pioneered research elucidating the functions of the SMN...

Journal: :Anales de pediatria 2015
A Madrid Rodríguez P L Martínez Martínez J M Ramos Fernández A Urda Cardona J Martínez Antón

OBJECTIVES To determine the incidence of spinal muscular atrophy (SMA) in our study population and genetic distribution and epidemiological and clinical characteristics and to analyze the level of care and development. MATERIAL AND METHOD Retrospective descriptive study of patients treated in our hospital in the past 25 years (from 1987 to early 2013), with a clinical and neurophysiological d...

2013
Dione T. Kobayashi Jing Shi Laurie Stephen Karri L. Ballard Ruth Dewey James Mapes Brett Chung Kathleen McCarthy Kathryn J. Swoboda Thomas O. Crawford Rebecca Li Thomas Plasterer Cynthia Joyce Wendy K. Chung Petra Kaufmann Basil T. Darras Richard S. Finkel Douglas M. Sproule William B. Martens Michael P. McDermott Darryl C. De Vivo Michael G. Walker Karen S. Chen

OBJECTIVES Spinal Muscular Atrophy (SMA) presents challenges in (i) monitoring disease activity and predicting progression, (ii) designing trials that allow rapid assessment of candidate therapies, and (iii) understanding molecular causes and consequences of the disease. Validated biomarkers of SMA motor and non-motor function would offer utility in addressing these challenges. Our objectives w...

Journal: :BMC Medicine 2009
Caterina Millino Marina Fanin Andrea Vettori Paolo Laveder Maria Luisa Mostacciuolo Corrado Angelini Gerolamo Lanfranchi

BACKGROUND Spinal muscular atrophy (SMA) is a neurodegenerative disorder associated with mutations of the survival motor neuron gene SMN and is characterized by muscle weakness and atrophy caused by degeneration of spinal motor neurons. SMN has a role in neurons but its deficiency may have a direct effect on muscle tissue. METHODS We applied microarray and quantitative real-time PCR to study ...

Journal: :Neuropathology and applied neurobiology 2014
Rachael A Powis Chantal A Mutsaers Thomas M Wishart Gillian Hunter Brunhilde Wirth Thomas H Gillingwater

AIM Levels of ubiquitin carboxyl-terminal hydrolase L1 (UCHL1) are robustly increased in spinal muscular atrophy (SMA) patient fibroblasts and mouse models. We therefore wanted to establish whether changes in UCHL1 contribute directly to disease pathogenesis, and to assess whether pharmacological inhibition of UCHL1 represents a viable therapeutic option for SMA. METHODS SMA mice and control ...

2012
Dhruv Sareen Allison D. Ebert Brittany M. Heins Jered V. McGivern Loren Ornelas Clive N. Svendsen

Spinal muscular atrophy (SMA) is a genetic disorder caused by a deletion of the survival motor neuron 1 gene leading to motor neuron loss, muscle atrophy, paralysis, and death. We show here that induced pluripotent stem cell (iPSC) lines generated from two Type I SMA subjects-one produced with lentiviral constructs and the second using a virus-free plasmid-based approach-recapitulate the diseas...

Journal: :Neuron 2005
Umrao R. Monani

Spinal muscular atrophy (SMA) is a neurodegenerative disease in humans and the most common genetic cause of infant mortality. The disease results in motor neuron loss and skeletal muscle atrophy. Despite a range of disease phenotypes, SMA is caused by mutations in a single gene, the Survival of Motor Neuron 1 (SMN1) gene. Recent advances have shed light on functions of the protein product of th...

Journal: :Human molecular genetics 2000
T Frugier F D Tiziano C Cifuentes-Diaz P Miniou N Roblot A Dierich M Le Meur J Melki

Deletion of the murine survival of motor neuron gene (SMN) exon 7, the most frequent mutation found in spinal muscular atrophy (SMA) patients, directed to neurons but not to skeletal muscle, enabled generation of a mouse model of SMA providing evidence that motor neurons are the primary target of the gene defect. Moreover, the mutated SMN protein (SMNDeltaC15) is dramatically reduced in the mot...

نمودار تعداد نتایج جستجو در هر سال

با کلیک روی نمودار نتایج را به سال انتشار فیلتر کنید