نتایج جستجو برای: smn gene

تعداد نتایج: 1142093  

Journal: :Human molecular genetics 2011
Elisa Dominguez Thibaut Marais Nicolas Chatauret Sofia Benkhelifa-Ziyyat Sandra Duque Philippe Ravassard Romain Carcenac Stéphanie Astord Aurélie Pereira de Moura Thomas Voit Martine Barkats

Spinal muscular atrophy (SMA) is the most common genetic disease leading to infant mortality. This neuromuscular disorder is caused by the loss or mutation of the telomeric copy of the 'survival of motor neuron' (Smn) gene, termed SMN1. Loss of SMN1 leads to reduced SMN protein levels, inducing degeneration of motor neurons (MN) and progressive muscle weakness and atrophy. To date, SMA remains ...

Journal: :The Biochemical journal 2005
Raphaël Rouget François Vigneault Circé Codio Camille Rochette Isabelle Paradis Régen Drouin Louise R Simard

There exist two SMN (survival motor neuron) genes in humans, the result of a 500 kb duplication in chromosome 5q13. Deletions/mutations in the SMN1 gene are responsible for childhood spinal muscular atrophy, an autosomal recessive neurodegenerative disorder. While the SMN1 and SMN2 genes are not functionally equivalent, up-regulation of the SMN2 gene represents an important therapeutic target. ...

Journal: :Chemistry & biology 2004
Mitchell R Lunn David E Root Allison M Martino Stephen P Flaherty Brian P Kelley Daniel D Coovert Arthur H Burghes Nguyen Thi Man Glenn E Morris Jianhua Zhou Elliot J Androphy Charlotte J Sumner Brent R Stockwell

Most patients with the pediatric neurodegenerative disease spinal muscular atrophy have a homozygous deletion of the survival motor neuron 1 (SMN1) gene, but retain one or more copies of the closely related SMN2 gene. The SMN2 gene encodes the same protein (SMN) but produces it at a low efficiency compared with the SMN1 gene. We performed a high-throughput screen of approximately 47,000 compoun...

Journal: :Human molecular genetics 1997
D D Coovert T T Le P E McAndrew J Strasswimmer T O Crawford J R Mendell S E Coulson E J Androphy T W Prior A H Burghes

The 38 kDa survival motor neuron (SMN) protein is encoded by two ubiquitously expressed genes: telomeric SMN (SMN(T)) and centromeric SMN (SMN(C)). Mutations in SMN(T), but not SMN(C), cause proximal spinal muscular atrophy (SMA), an autosomal recessive disorder that results in loss of motor neurons. SMN is found in the cytoplasm and nucleus. The nuclear form is located in structures termed gem...

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: :Human molecular genetics 2015
Naresh K Genabai Saif Ahmad Zhanying Zhang Xiaoting Jiang Cynthia A Gabaldon Laxman Gangwani

Mutation of the Survival Motor Neuron 1 (SMN1) gene causes spinal muscular atrophy (SMA), an autosomal recessive neurodegenerative disorder that occurs in early childhood. Degeneration of spinal motor neurons caused by SMN deficiency results in progressive muscle atrophy and death in SMA. The molecular mechanism underlying neurodegeneration in SMA is unknown. No treatment is available to preven...

Journal: :Human molecular genetics 2011
Lisa Hubers Hector Valderrama-Carvajal Janik Laframboise Janie Timbers Gabriel Sanchez Jocelyn Côté

Spinal muscular atrophy is an autosomal-recessive neuromuscular disease caused by disruption of the survival of motor neuron (SMN) gene, which promotes cytoplasmic assembly of the splicing core machinery. It remains unclear how a deficiency in SMN results in a disorder leading to selective degeneration of lower motor neurons. We report here that SMN interacts with RNA-binding protein HuD in neu...

2013
Rebecca Borg Ruben J. Cauchi

Membership of the survival motor neuron (SMN) complex extends to nine factors, including the SMN protein, the product of the spinal muscular atrophy (SMA) disease gene, Gemins 2-8 and Unrip. The best-characterised function of this macromolecular machine is the assembly of the Sm-class of uridine-rich small nuclear ribonucleoprotein (snRNP) particles and each SMN complex member has a key role du...

Journal: :Human molecular genetics 2009
Kum-Loong Boon Shu Xiao Michelle L McWhorter Thomas Donn Emma Wolf-Saxon Markus T Bohnsack Cecilia B Moens Christine E Beattie

Spinal muscular atrophy (SMA), a recessive genetic disease, affects lower motoneurons leading to denervation, atrophy, paralysis and in severe cases death. Reduced levels of survival motor neuron (SMN) protein cause SMA. As a first step towards generating a genetic model of SMA in zebrafish, we identified three smn mutations. Two of these alleles, smnY262stop and smnL265stop, were stop mutation...

Journal: :Journal of cell science 2006
Benoît Renvoisé Kevinee Khoobarry Marie-Claude Gendron Christian Cibert Louis Viollet Suzie Lefebvre

Mutations of the survival motor neuron gene SMN1 cause the inherited disease spinal muscular atrophy (SMA). The ubiquitous SMN protein facilitates the biogenesis of spliceosomal small nuclear ribonucleoproteins (snRNPs). The protein is detected in the cytoplasm, nucleoplasm and enriched with snRNPs in nuclear Cajal bodies. It is structurally divided into at least an amino-terminal region rich i...

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