نتایج جستجو برای: ژن smn1

تعداد نتایج: 16245  

Journal: :Current Biology 2001
Michael P Terns Rebecca M Terns

Recent studies indicate that the protein affected in spinal muscular atrophy, SMN, plays a role in the assembly of a number of macromolecular complexes that function in the nucleus, interacting with its partner proteins via their arginine- and glycine-rich domains.

2013
Paolo d’Errico Denise Locatelli Silvia Capra Adele Finardi Francesca Colciaghi Veronica Setola Mineko Terao Enrico Garattini

The axonal SMN (a-SMN) protein is a truncated isoform of SMN1, the spinal muscular atrophy (SMA) disease gene. a-SMN is selectively localized in axons and endowed with remarkable axonogenic properties. At present, the role of a-SMN in SMA is unknown. As a first step to verify a link between a-SMN and SMA, we investigated by means of over-expression experiments in NSC34 motor neurons whether SMA...

2013
C. Lebrero

Spinal muscular atrophy (SMA) is a recessive disorder involving the degradation and subsequent loss of motor neurons from the spinal cord. The main cause of SMA is related with the absence of the functional form of the survival motor neuron (SMN) gene (SMN1). This gene presents a telomeric (SMN1) and a centromeric (SMN2) copies that differ in 5 nucleotides. This mutation produces an alternative...

Journal: :Human molecular genetics 2012
Melissa Osborne Daniel Gomez Zhihua Feng Corissa McEwen Jose Beltran Kim Cirillo Bassem El-Khodor Ming-Yi Lin Yun Li Wendy M Knowlton David D McKemy Laurent Bogdanik Katherine Butts-Dehm Kimberly Martens Crystal Davis Rosalinda Doty Keegan Wardwell Afshin Ghavami Dione Kobayashi Chien-Ping Ko Sylvie Ramboz Cathleen Lutz

A number of mouse models for spinal muscular atrophy (SMA) have been genetically engineered to recapitulate the severity of human SMA by using a targeted null mutation at the mouse Smn1 locus coupled with the transgenic addition of varying copy numbers of human SMN2 genes. Although this approach has been useful in modeling severe SMA and very mild SMA, a mouse model of the intermediate form of ...

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: :Molecular and cellular biology 2006
Nirmal K Singh Natalia N Singh Elliot J Androphy Ravindra N Singh

Humans have two nearly identical copies of the Survival Motor Neuron (SMN) gene, SMN1 and SMN2. In spinal muscular atrophy (SMA), SMN2 is not able to compensate for the loss of SMN1 due to exclusion of exon 7. Here we describe a novel inhibitory element located immediately downstream of the 5' splice site in intron 7. We call this element intronic splicing silencer N1 (ISS-N1). Deletion of ISS-...

Journal: :Human molecular genetics 2000
N Owen C L Doe J Mellor K E Davies

Childhood onset spinal muscular atrophy (SMA) is a common autosomal recessive disorder primarily characterized by the loss of lower alpha motor neurons. The underlying chromosomal defects causing SMA have been found in the survival motor neuron (SMN) gene. SMN has been shown previously to play a role in both snRNP biogenesis and mRNA processing, although direct evidence for the relationship bet...

Journal: :Human molecular genetics 1999
T Vitali V Sossi F Tiziano S Zappata A Giuli M Paravatou-Petsotas G Neri C Brahe

Spinal muscular atrophy (SMA) is a common autosomal recessive neuromuscular disorder which presents with various clinical phenotypes ranging from severe to very mild. All forms are caused by the homozygous absence of the survival motor neuron ( SMN1 ) gene. SMN1 and a nearly identical copy ( SMN2 ) are located in a duplicated region at 5q13 and encode identical proteins. The genetic basis for t...

Journal: :Human molecular genetics 2013
Melissa S Cobb Ferril F Rose Hansjörg Rindt Jacqueline J Glascock Monir Shababi Madeline R Miller Erkan Y Osman Pei-Fen Yen Michael L Garcia Brittanie R Martin Mary J Wetz Chiara Mazzasette Zhihua Feng Chien-Ping Ko Christian L Lorson

Spinal Muscular Atrophy (SMA) is due to the loss of the survival motor neuron gene 1 (SMN1), resulting in motor neuron (MN) degeneration, muscle atrophy and loss of motor function. While SMN2 encodes a protein identical to SMN1, a single nucleotide difference in exon 7 causes most of the SMN2-derived transcripts to be alternatively spliced resulting in a truncated and unstable protein (SMNΔ7). ...

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