نتایج جستجو برای: survival motor neuron smn gene

تعداد نتایج: 1595688  

Journal: :Human molecular genetics 2011
Elisa Dominguez Thibaut Marais Nicolas Chatauret Sofia Benkhelifa-Ziyyat Sandra Duque Philippe Ravassard Romain Carcenac Stéphanie Astord Aurélie Pereira de Moura Thomas Voit Martine Barkats

Spinal muscular atrophy (SMA) is the most common genetic disease leading to infant mortality. This neuromuscular disorder is caused by the loss or mutation of the telomeric copy of the 'survival of motor neuron' (Smn) gene, termed SMN1. Loss of SMN1 leads to reduced SMN protein levels, inducing degeneration of motor neurons (MN) and progressive muscle weakness and atrophy. To date, SMA remains ...

1999
Bernard Charroux Livio Pellizzoni Robert A. Perkinson Andrej Shevchenko Matthias Mann Gideon Dreyfuss

The survival of motor neurons ( SMN ) gene is the disease gene of spinal muscular atrophy (SMA), a common motor neuron degenerative disease. The SMN protein is part of a complex containing several proteins, of which one, SIP1 (SMN interacting protein 1), has been characterized so far. The SMN complex is found in both the cytoplasm and in the nucleus, where it is concentrated in bodies called ge...

Journal: :Archives of neurology 1999
J S Parboosingh V Meininger D McKenna-Yasek R H Brown G A Rouleau

BACKGROUND Amyotrophic lateral sclerosis (ALS) is a rapidly progressive, invariably lethal disease resulting from the premature death of motor neurons of the motor cortex, brainstem, and spinal cord. In approximately 15% of familial ALS cases, the copper/zinc superoxide dismutase gene is mutated; a juvenile form of familial ALS has been linked to chromosome 2. No cause has been identified in th...

2016
Xin Zhao Zhihua Feng Karen K. Y. Ling Anna Mollin Josephine Sheedy Shirley Yeh Janet Petruska Jana Narasimhan Amal Dakka Ellen M. Welch Gary Karp Karen S. Chen Friedrich Metzger Hasane Ratni Francesco Lotti Sarah Tisdale Nikolai A. Naryshkin Livio Pellizzoni Sergey Paushkin Chien-Ping Ko Marla Weetall

Spinal muscular atrophy (SMA) is caused by the loss or mutation of both copies of the survival motor neuron 1 (SMN1) gene. The related SMN2 gene is retained, but due to alternative splicing of exon 7, produces insufficient levels of the SMN protein. Here, we systematically characterize the pharmacokinetic and pharmacodynamics properties of the SMN splicing modifier SMN-C1. SMN-C1 is a low-molec...

2016
Ivan Gallotta Nadia Mazzarella Alessandra Donato Alessandro Esposito Justin C. Chaplin Silvana Castro Giuseppina Zampi Giorgio S. Battaglia Massimo A. Hilliard Paolo Bazzicalupo Elia Di Schiavi

Spinal muscular atrophy is a devastating disease that is characterized by degeneration and death of a specific subclass of motor neurons in the anterior horn of the spinal cord. Although the gene responsible, survival motor neuron 1 (SMN1), was identified 20 years ago, it has proven difficult to investigate its effects in vivo. Consequently, a number of key questions regarding the molecular and...

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...

2016
Mehdi Eshraghi Emily McFall Sabrina Gibeault Rashmi Kothary

Spinal muscular atrophy (SMA) is caused by mutations or deletions in the Survival Motor Neuron 1 (SMN1) gene in humans. Modifiers of the SMA symptoms have been identified and genetic background has a substantial effect in the phenotype and survival of the severe mouse model of SMA. Previously, we generated the less severe Smn2B/- mice on a mixed genetic background. To assess the phenotype of Sm...

Journal: :Neuroscience 2011
R S Anderton B P Meloni F L Mastaglia W K Greene S Boulos

Spinal muscular atrophy (SMA), a neurodegenerative disorder primarily affecting motor neurons, is the most common genetic cause of infant death. This incurable disease is caused by the absence of a functional SMN1 gene and a reduction in full length survival of motor neuron (SMN) protein. In this study, a neuroprotective function of SMN was investigated in differentiated human SH-SY5Y cells usi...

2012
Li-Kai Tsai

Spinal muscular atrophy (SMA) is an autosomal recessive neurodegenerative disorder, leading to progressive muscle weakness, atrophy, and sometimes premature death. SMA is caused by mutation or deletion of the survival motor neuron-1 (SMN1) gene. An effective treatment does not presently exist. Since the severity of the SMA phenotype is inversely correlated with expression levels of SMN, the SMN...

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