نتایج جستجو برای: survival motor neuron gene

تعداد نتایج: 1595196  

Journal: :Human molecular genetics 2011
Marta Bosch-Marcé Claribel D Wee Tara L Martinez Celeste E Lipkes Dong W Choe Lingling Kong James P Van Meerbeke Antonio Musarò Charlotte J Sumner

Spinal muscular atrophy (SMA) is an inherited motor neuron disease caused by the mutation of the survival motor neuron 1 (SMN1) gene and deficiency of the SMN protein. Severe SMA mice have abnormal motor function and small, immature myofibers early in development suggesting that SMN protein deficiency results in retarded muscle growth. Insulin-like growth factor 1 (IGF-1) stimulates myoblast pr...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2017
Mohini Jangi Christina Fleet Patrick Cullen Shipra V Gupta Shila Mekhoubad Eric Chiao Norm Allaire C Frank Bennett Frank Rigo Adrian R Krainer Jessica A Hurt John P Carulli John F Staropoli

Spinal muscular atrophy (SMA), an autosomal recessive neuromuscular disease, is the leading monogenic cause of infant mortality. Homozygous loss of the gene survival of motor neuron 1 (SMN1) causes the selective degeneration of lower motor neurons and subsequent atrophy of proximal skeletal muscles. The SMN1 protein product, survival of motor neuron (SMN), is ubiquitously expressed and is a key...

Journal: :The Journal of biological chemistry 2002
Philip J Young Patricia M Day Jianhua Zhou Elliot J Androphy Glenn E Morris Christian L Lorson

Mutations in the SMN1 (survival motor neuron 1) gene cause spinal muscular atrophy (SMA). We now show that SMN protein, the SMN1 gene product, interacts directly with the tumor suppressor protein, p53. Pathogenic missense mutations in SMN reduce both self-association and p53 binding by SMN, and the extent of the reductions correlate with disease severity. The inactive, truncated form of SMN pro...

2011
James N. Sleigh Thomas H. Gillingwater Kevin Talbot

Spinal muscular atrophy (SMA), which is caused by inactivating mutations in the survival motor neuron 1 (SMN1) gene, is characterized by loss of lower motor neurons in the spinal cord. The gene encoding SMN is very highly conserved in evolution, allowing the disease to be modeled in a range of species. The similarities in anatomy and physiology to the human neuromuscular system, coupled with th...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2005
Christian S Lobsiger Michael L Garcia Christopher M Ward Don W Cleveland

Eliminating assembled neurofilaments (NFs) from axons or misaccumulating NFs in motor neuron cell bodies strongly slows disease in mouse models of mutant superoxide dismutase 1 (SOD1)-induced amyotrophic lateral sclerosis. One proposal for how reducing axonal NFs can increase survival is that the multiphosphorylated tail domains of the two larger NF subunits act in motor neuron cell bodies as p...

Journal: :Molecular and cellular biology 2005
Laxman Gangwani Richard A Flavell Roger J Davis

Mutation of the survival motor neurons 1 (SMN1) gene causes motor neuron apoptosis and represents the major cause of spinal muscular atrophy in humans. Biochemical studies have established that the SMN protein plays an important role in spliceosomal small nuclear ribonucleoprotein (snRNP) biogenesis and that the SMN complex can interact with the zinc finger protein ZPR1. Here we report that tar...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2000
D A Kerr J P Nery R J Traystman B N Chau J M Hardwick

Spinal muscular atrophy (SMA) is attributed to mutations in the SMN1 gene, leading to loss of spinal cord motor neurons. The neurotropic Sindbis virus vector system was used to investigate a role for the survival motor neuron (SMN) protein in regulating neuronal apoptosis. Here we show that SMN protects primary neurons and differentiated neuron-like stem cells, but not cultured cell lines from ...

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