نتایج جستجو برای: smn gene

تعداد نتایج: 1142093  

Journal: :Journal of neuropathology and experimental neurology 2006
Alessio Giavazzi Veronica Setola Alessandro Simonati Giorgio Battaglia

Despite recent data on the cellular function of the survival motor neuron (SMN) gene, the spinal muscular atrophy (SMA) disease gene, the role of the SMN protein in motor neurons and hence in the pathogenesis of SMA is still unclear. The spatial and temporal expression of SMN in neurons, particularly during development, could help in verifying the hypotheses on the SMN protein functions so far ...

Journal: :Human molecular genetics 2009
Eileen Workman Luciano Saieva Tessa L Carrel Thomas O Crawford Don Liu Cathleen Lutz Christine E Beattie Livio Pellizzoni Arthur H M Burghes

Spinal muscular atrophy (SMA) is an autosomal recessive neurodegenerative disease. Loss of the survival motor neuron (SMN1) gene, in the presence of the SMN2 gene causes SMA. SMN functions in snRNP assembly in all cell types, however, it is unclear how this function results in specifically motor neuron cell death. Lack of endogenous mouse SMN (Smn) in mice results in embryonic lethality. Introd...

2009
Michael Briese Behrooz Esmaeili Sandrine Fraboulet Emma C. Burt Stefanos Christodoulou Paula R. Towers Kay E. Davies David B. Sattelle

Spinal muscular atrophy is the most common genetic cause of infant mortality and is characterized by degeneration of lower motor neurons leading to muscle wasting. The causative gene has been identified as survival motor neuron (SMN). The invertebrate model organism Caenorhabditis elegans contains smn-1, the ortholog of human SMN. Caenorhabditis elegans smn-1 is expressed in various tissues inc...

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...

Journal: :Human molecular genetics 2013
Faraz Farooq Francisco Abadía-Molina Duncan MacKenzie Jeremiah Hadwen Fahad Shamim Sean O'Reilly Martin Holcik Alex MacKenzie

The loss of functional Survival Motor Neuron (SMN) protein due to mutations or deletion in the SMN1 gene causes autosomal recessive neurodegenerative spinal muscle atrophy (SMA). A potential treatment strategy for SMA is to upregulate the amount of SMN protein originating from the highly homologous SMN2 gene, compensating in part for the absence of the functional SMN1 gene. We have previously s...

Journal: :Human molecular genetics 1996
C Brahe O Clermont S Zappata F Tiziano J Melki G Neri

Recently, a spinal muscular atrophy (SMA) determining gene, termed survival motor neuron (SMN) gene, has been isolated from the 5q13 region and found deleted in most patients. A highly homologous copy of this gene has also been isolated and located in a centromeric position. We have analyzed 158 patients (SMA types I-IV) and found deletions of SMN exon 7 in 96.8%. Mutations other than gross del...

Journal: :Molecular and cellular biology 2009
Barrington G Burnett Eric Muñoz Animesh Tandon Deborah Y Kwon Charlotte J Sumner Kenneth H Fischbeck

Spinal muscular atrophy (SMA) is caused by mutations of the survival of motor neuron (SMN1) gene and deficiency of full-length SMN protein (FL-SMN). All SMA patients retain one or more copies of the SMN2 gene, but the principal protein product of SMN2 lacks exon 7 (SMNDelta7) and is unable to compensate for a deficiency of FL-SMN. SMN is known to oligomerize and form a multimeric protein comple...

2013
Jonathan J Cherry Erkan Y Osman Matthew C Evans Sungwoon Choi Xuechao Xing Gregory D Cuny Marcie A Glicksman Christian L Lorson Elliot J Androphy

Spinal muscular atrophy (SMA) is a neurodegenerative disease that causes progressive muscle weakness, which primarily targets proximal muscles. About 95% of SMA cases are caused by the loss of both copies of the SMN1 gene. SMN2 is a nearly identical copy of SMN1, which expresses much less functional SMN protein. SMN2 is unable to fully compensate for the loss of SMN1 in motor neurons but does p...

2010
Salah Mahmoudi Sofia Henriksson Irene Weibrecht Stephen Smith Ola Söderberg Staffan Strömblad Klas G. Wiman Marianne Farnebo

The WRAP53 gene gives rise to a p53 antisense transcript that regulates p53. This gene also encodes a protein that directs small Cajal body-specific RNAs to Cajal bodies. Cajal bodies are nuclear organelles involved in diverse functions such as processing ribonucleoproteins important for splicing. Here we identify the WRAP53 protein as an essential factor for Cajal body maintenance and for dire...

Journal: :Human molecular genetics 2002
Wilfried Rossoll Ann-Kathrin Kröning Uta-Maria Ohndorf Clemens Steegborn Sibylle Jablonka Michael Sendtner

Spinal muscular atrophy (SMA), the most common hereditary motor neuron disease in children and young adults is caused by mutations in the telomeric survival motor neuron (SMN1) gene. The human genome, in contrast to mouse, contains a second SMN gene (SMN2) which codes for a gene product which is alternatively spliced at the C-terminus, but also gives rise to low levels of full-length SMN protei...

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