نتایج جستجو برای: smn2 protein

تعداد نتایج: 1234838  

Journal: :Human molecular genetics 2012
Thomas W Bebee Catherine E Dominguez Somayeh Samadzadeh-Tarighat Kristi L Akehurst Dawn S Chandler

Spinal muscular atrophy (SMA) is a progressive neurodegenerative disease associated with low levels of the essential survival motor neuron (SMN) protein. Reduced levels of SMN is due to the loss of the SMN1 gene and inefficient splicing of the SMN2 gene caused by a C>T mutation in exon 7. Global analysis of the severe SMNΔ7 SMA mouse model revealed altered splicing and increased levels of the h...

2014
Ahmed Moursy Frédéric H.-T. Allain Antoine Cléry

Regulation of SMN2 exon 7 splicing is crucial for the production of active SMN protein and the survival of Spinal Muscular Atrophy (SMA) patients. One of the most efficient activators of exon 7 inclusion is hnRNP G, which is recruited to the exon by Tra2-β1. We report that in addition to the C-terminal region of hnRNP G, the RNA Recognition Motif (RRM) and the middle part of the protein contain...

2014
Modibo Sangaré Brant Hendrickson Hammadoun Ali Sango Kelian Chen Jonathan Nofziger Abdelbasset Amara Amalia Dutra Alice B. Schindler Aldiouma Guindo Mahamadou Traoré George Harmison Evgenia Pak Fatoumata N'Go Yaro Katherine Bricceno Christopher Grunseich Guibin Chen Manfred Boehm Kristen Zukosky Nouhoum Bocoum Katherine G. Meilleur Fatoumata Daou Koumba Bagayogo Yaya Ibrahim Coulibaly Mahamadou Diakité Michael P. Fay Hee‐Suk Lee Ali Saad Moez Gribaa Andrew B. Singleton Youssoufa Maiga Sungyoung Auh Guida Landouré Rick M. Fairhurst Barrington G. Burnett Thomas Scholl Kenneth H. Fischbeck

OBJECTIVE Spinal muscular atrophy (SMA) is one of the most common severe hereditary diseases of infancy and early childhood in North America, Europe, and Asia. SMA is usually caused by deletions of the survival motor neuron 1 (SMN1) gene. A closely related gene, SMN2, modifies the disease severity. SMA carriers have only 1 copy of SMN1 and are relatively common (1 in 30-50) in populations of Eu...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2003
Leigh A Skordis Matthew G Dunckley Baigong Yue Ian C Eperon Francesco Muntoni

The multiplicity of proteins compared with genes in mammals owes much to alternative splicing. Splicing signals are so subtle and complex that small perturbations may allow the production of new mRNA variants. However, the flexibility of splicing can also be a liability, and several genetic diseases result from single-base changes that cause exons to be skipped during splicing. Conventional oli...

Journal: :Journal of neuropathology and experimental neurology 2015
Brian N Harding Shingo Kariya Umrao R Monani Wendy K Chung Maryjane Benton Sabrina W Yum Gihan Tennekoon Richard S Finkel

Neuropathologic findings within the central and peripheral nervous systems in patients with spinal muscular atrophy type I (SMA-I) were examined in relation to genetic, clinical, and electrophysiologic features. Five infants representing the full clinical spectrum of SMA-I were examined clinically for compound motor action potential amplitude and SMN2 gene copy number; morphologic analyses of p...

2013
Kentaro Sahashi Karen K Y Ling Yimin Hua John Erby Wilkinson Tomoki Nomakuchi Frank Rigo Gene Hung David Xu Ya-Ping Jiang Richard Z Lin Chien-Ping Ko C Frank Bennett Adrian R Krainer

Loss-of-function mutations in SMN1 cause spinal muscular atrophy (SMA), a leading genetic cause of infant mortality. The related SMN2 gene expresses suboptimal levels of functional SMN protein, due to a splicing defect. Many SMA patients reach adulthood, and there is also adult-onset (type IV) SMA. There is currently no animal model for adult-onset SMA, and the tissue-specific pathogenesis of p...

Journal: :Frontiers in bioscience 2010
Thomas W Bebee Jordan T Gladman Dawn S Chandler

Proximal spinal muscular atrophy (SMA) is a neuromuscular disease caused by low levels of the survival motor neuron (SMN) protein. The reduced SMN levels are due to loss of the survival motor neuron-1 (SMN1) gene. Humans carry a nearly identical SMN2 gene that generates a truncated protein, due to a C to T nucleotide alteration in exon 7 that leads to inefficient RNA splicing of exon 7. This ex...

2015
Nur Imma Fatimah Harahap Dian Kesumapramudya Nurputra Mawaddah Ar Rochmah Ai Shima Naoya Morisada Toru Takarada Atsuko Takeuchi Yumi Tohyama Shinichiro Yanagisawa Hisahide Nishio

Spinal muscular atrophy (SMA) is a common autosomal recessive neuromuscular disorder that is currently incurable. SMA is caused by decreased levels of the survival motor neuron protein (SMN), as a result of loss or mutation of SMN1. Although the SMN1 homolog SMN2 also produces some SMN protein, it does not fully compensate for the loss or dysfunction of SMN1. Salbutamol, a β2-adrenergic recepto...

2009
Till Geib Klemens J. Hertel

BACKGROUND Spinal Muscular Atrophy (SMA) is an autosomal recessive disease that leads to specific loss of motor neurons. It is caused by deletions or mutations of the survival of motor neuron 1 gene (SMN1). The remaining copy of the gene, SMN2, generates only low levels of the SMN protein due to a mutation in SMN2 exon 7 that leads to exon skipping. METHODOLOGY/PRINCIPAL FINDINGS To correct S...

Journal: :Genes & development 2010
Yimin Hua Kentaro Sahashi Gene Hung Frank Rigo Marco A Passini C Frank Bennett Adrian R Krainer

Increasing survival of motor neuron 2, centromeric (SMN2) exon 7 inclusion to express more full-length SMN protein in motor neurons is a promising approach to treat spinal muscular atrophy (SMA), a genetic neurodegenerative disease. Previously, we identified a potent 2'-O-(2-methoxyethyl) (MOE) phosphorothioate-modified antisense oligonucleotide (ASO) that blocks an SMN2 intronic splicing silen...

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