نتایج جستجو برای: survival motor neuron smn gene

تعداد نتایج: 1595688  

Journal: :Molecular human reproduction 1998
J C Dreesen M Bras C de Die-Smulders J C Dumoulin J M Cobben J L Evers H J Smeets J P Geraedts

After Duchenne muscular dystrophy, spinal muscular atrophy (SMA) is the most common severe neuromuscular disease in childhood. Since 1995, homozygous deletions in exon 7 of the survival motor neuron (SMN) gene have been described in >90-95% of SMA patients. However, the presence of a highly homologous SMN copy gene complicates the detection of exon 7 deletions. This paper describes the adjustme...

Journal: :The Journal of biological chemistry 2002
Livio Pellizzoni Jennifer Baccon Juri Rappsilber Matthias Mann Gideon Dreyfuss

The survival of motor neurons (SMN) protein, the product of the gene responsible for the motor neuron degenerative disease spinal muscular atrophy (SMA), is part of a large macromolecular complex. The SMN complex is localized in both the cytoplasm and the nucleus and contains SMN, Gemin2, Gemin3, Gemin4, Gemin5, and a few not yet identified proteins. The SMN complex plays a key role in the biog...

Journal: :Neuron 2017
Constantin d’Ydewalle Daniel M. Ramos Noah J. Pyles Shi-Yan Ng Mariusz Gorz Celeste M. Pilato Karen Ling Lingling Kong Amanda J. Ward Lee L. Rubin Frank Rigo C. Frank Bennett Charlotte J. Sumner

The neuromuscular disorder spinal muscular atrophy (SMA), the most common inherited killer of infants, is caused by insufficient expression of survival motor neuron (SMN) protein. SMA therapeutics development efforts have focused on identifying strategies to increase SMN expression. We identified a long non-coding RNA (lncRNA) that arises from the antisense strand of SMN, SMN-AS1, which is enri...

Journal: :The Journal of biological chemistry 2001
K W Jones K Gorzynski C M Hales U Fischer F Badbanchi R M Terns M P Terns

Disruption of the survival motor neuron (SMN) gene leads to selective loss of spinal motor neurons, resulting in the fatal human neurodegenerative disorder spinal muscular atrophy (SMA). SMN has been shown to function in spliceosomal small nuclear ribonucleoprotein (snRNP) biogenesis and pre-mRNA splicing. We have demonstrated that SMN also interacts with fibrillarin, a highly conserved nucleol...

2012
Chen-Hung Ting Hsin-Lan Wen Hui-Chun Liu Hsiu-Mei Hsieh-Li Hung Li Sue Lin-Chao

Proximal spinal muscular atrophy (SMA) is a neurodegenerative disorder caused by deficiency of the ubiquitous Survival of Motor Neuron (SMN) protein. SMN has been shown to be transported in granules along the axon and moved through cytoskeletal elements. However, the role and nature of SMN granules are still not well characterized. Here, using immunocytochemical methods and time-lapse studies w...

Journal: :Cell 2008
Zhenxi Zhang Francesco Lotti Kimberly Dittmar Ihab Younis Lili Wan Mumtaz Kasim Gideon Dreyfuss

The survival of motor neurons (SMN) protein is essential for the biogenesis of small nuclear RNA (snRNA)-ribonucleoproteins (snRNPs), the major components of the pre-mRNA splicing machinery. Though it is ubiquitously expressed, SMN deficiency causes the motor neuron degenerative disease spinal muscular atrophy (SMA). We show here that SMN deficiency, similar to that which occurs in severe SMA, ...

Journal: :Molecular therapy : the journal of the American Society of Gene Therapy 2005
Csilla Madocsai Sharlene R Lim Till Geib Bianca J Lam Klemens J Hertel

Mutations in one of the duplicated survival of motor neuron (SMN) genes lead to the progressive loss of motor neurons and subsequent development of spinal muscular atrophy (SMA), a common, and usually fatal, hereditary disease. Homozygous absence of the telomeric copy (SMN1) correlates with development of SMA because differential splicing of the centromeric copy (SMN2) leads to exon 7 skipping ...

Journal: :Cell 1998
Livio Pellizzoni Naoyuki Kataoka Bernard Charroux Gideon Dreyfuss

Spinal muscular atrophy (SMA) is a common motor neuron degenerative disease that results from reduced levels of, or mutations in, the Survival of Motor Neurons (SMN) protein. SMN is found in the cytoplasm and the nucleus where it is concentrated in gems. SMN interacts with spliceosomal snRNP proteins and is critical for snRNP assembly in the cytoplasm. We show that a dominant-negative mutant SM...

2016
Modibo Sangare Ilo Dicko Cheick Oumar Guinto Adama Sissoko Kekouta Dembele Youlouza Coulibaly Siaka Y. Coulibaly Guida Landoure Abdallah Diallo Mamadou Dolo Housseini Dolo Boubacar Maiga Moussa Traore Mamadou Karembe Kadiatou Traore Amadou Toure Mariam Sylla Arouna Togora Souleymane Coulibaly Sékou Fantamady Traore Brant Hendrickson Katherine Bricceno Alice B. Schindler Angela Kokkinis Katherine G. Meilleur Hammadoun Ali Sangho Brehima Diakite Yaya Kassogue Yaya Ibrahim Coulibaly Barrington Burnett Youssoufa Maiga Seydou Doumbia Kenneth H. Fischbeck

Introduction Spinal muscular atrophy (SMA) and sporadic amyotrophic lateral sclerosis (SALS) are both motor neuron disorders. SMA results from the deletion of the survival motor neuron (SMN) 1 gene. High or low SMN1 copy number and the absence of SMN2 have been reported as risk factors for the development or severity of SALS. Objective To investigate the role of SMN gene copy number in the on...

Journal: :Human molecular genetics 2010
Mélissa Bowerman Ariane Beauvais Carrie L Anderson Rashmi Kothary

Spinal muscular atrophy (SMA) is an inherited disease resulting in the highest mortality of children under the age of two. SMA is caused by mutations or deletions in the survival motor neuron 1 (SMN1) gene, leading to aberrant neuromuscular junction (NMJ) development and the loss of spinal cord alpha-motor neurons. Here, we show that Smn depletion leads to increased activation of RhoA, a major ...

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