نتایج جستجو برای: survival motor neuron gene

تعداد نتایج: 1595196  

Journal: :Cell 1995
Suzie Lefebvre Lydie Bürglen Sophie Reboullet Olivier Clermont Philippe Burlet Louis Viollet Bernard Benichou Corinne Cruaud Philippe Millasseau Massimo Zeviani Denis Le Paslier Jean Frézal Daniel Cohen Jean Weissenbach Arnold Munnich Judith Melki

Spinal muscular atrophy (SMA) is a common fatal autosomal recessive disorder characterized by degeneration of lower motor neurons, leading to progressive paralysis with muscular atrophy. The gene for SMA has been mapped to chromosome 5q13, where large-scale deletions have been reported. We describe here the inverted duplication of a 500 kb element in normal chromosomes and narrow the critical r...

Journal: :Human molecular genetics 2011
Deborah Y Kwon William W Motley Kenneth H Fischbeck Barrington G Burnett

Spinal muscular atrophy (SMA) is a neuromuscular disorder caused by reduced levels of the survival motor neuron (SMN) protein. Here we show that the proteasome inhibitor, bortezomib, increases SMN in cultured cells and in peripheral tissues of SMA model mice. Bortezomib-treated animals had improved motor function, which was associated with reduced spinal cord and muscle pathology and improved n...

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2006
Alvaro G Estévez Mary Anne Sahawneh Philipp S Lange Narae Bae Mariela Egea Rajiv R Ratan

When deprived of trophic factors, the majority of cultured motor neurons undergo nitric oxide-dependent apoptosis. However, for reasons that have remained unclear, 30-50% of the motor neurons survive for several days without trophic factors. Here we hypothesize that the resistance of this motor neuron subpopulation to trophic factor deprivation can be attributed to diminished nitric oxide produ...

Journal: :Human molecular genetics 2014
James N Sleigh Antón Barreiro-Iglesias Peter L Oliver Angeliki Biba Thomas Becker Kay E Davies Catherina G Becker Kevin Talbot

Spinal muscular atrophy (SMA) is characterized by the selective loss of spinal motor neurons owing to reduced levels of survival motor neuron (Smn) protein. In addition to its well-established role in assembling constituents of the spliceosome, diverse cellular functions have been proposed for Smn, but the reason why low levels of this widely expressed protein result in selective motor neuron p...

Journal: :Hong Kong medical journal = Xianggang yi xue za zhi 2001
P J Shaw

Scientific evidence is emerging to indicate that motor neuron injury in motor neuron disease may reflect a complex interplay between genetic factors, oxidative stress, and imbalance of the glutamatergic excitatory control of motor neurons, which may result in damage to critical target proteins and organelles. The relative importance of these factors is likely to vary in different subgroups of p...

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