Literature DB >> 9302277

Expression of the SMN gene, the spinal muscular atrophy determining gene, in the mammalian central nervous system.

G Battaglia1, A Princivalle, F Forti, C Lizier, M Zeviani.   

Abstract

The survival motor neuron (SMN) gene is the putative disease gene for human spinal muscular atrophy (SMA), an autosomal recessive disorder characterized by progressive degeneration of lower motor neurons. Two copies of the gene, centromeric and telomeric, are present in the same 5q13 chromosomal region in humans. However, only the telomeric gene is affected in SMA. The SMN gene(s) encode(s) a novel protein of unknown function. To gain insights into the role of SMN in neurons, we have identified the SMN gene ortholog in the rat, and investigated SMN expression in the CNS of rat, monkey and humans by immunocytochemistry and in situ hybridization experiments. Antibodies against the SMN amino-terminus specifically recognized a single protein identical to the in vitro translation products of human and rat SMN cDNAs. The SMN gene transcript and product were widely but unevenly expressed throughout cerebral and spinal cord areas. The SMN protein was localized mainly in the cytoplasm of specific neuronal systems, and it was particularly expressed in lower motor neurons of newborn and adult animals. Likewise, a strong hybridization signal was detected in lamina IX of the spinal ventral horn. These results support the relevance of SMN for the motor neuron function and the pathogenetic role of the SMN gene in the neuronal degeneration associated with SMA.

Entities:  

Mesh:

Substances:

Year:  1997        PMID: 9302277     DOI: 10.1093/hmg/6.11.1961

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  37 in total

Review 1.  Regulation of alternative RNA splicing by exon definition and exon sequences in viral and mammalian gene expression.

Authors:  Zhi-Ming Zheng
Journal:  J Biomed Sci       Date:  2004 May-Jun       Impact factor: 8.410

Review 2.  Applicability of histone deacetylase inhibition for the treatment of spinal muscular atrophy.

Authors:  Sebastian Lunke; Assam El-Osta
Journal:  Neurotherapeutics       Date:  2013-10       Impact factor: 7.620

3.  Radiographic and Respiratory Effects of Growing Rods in Children With Spinal Muscular Atrophy.

Authors:  Rachel L Lenhart; Sylvester Youlo; Mary K Schroth; Kenneth J Noonan; James McCarthy; David Mann; Scott Hetzel; Sarah A Sund; Matthew A Halanski
Journal:  J Pediatr Orthop       Date:  2017-12       Impact factor: 2.324

4.  Gene activation of SMN by selective disruption of lncRNA-mediated recruitment of PRC2 for the treatment of spinal muscular atrophy.

Authors:  Caroline J Woo; Verena K Maier; Roshni Davey; James Brennan; Guangde Li; John Brothers; Brian Schwartz; Susana Gordo; Anne Kasper; Trevor R Okamoto; Hans E Johansson; Berhan Mandefro; Dhruv Sareen; Peter Bialek; B Nelson Chau; Balkrishen Bhat; David Bullough; James Barsoum
Journal:  Proc Natl Acad Sci U S A       Date:  2017-02-13       Impact factor: 11.205

5.  The Antisense Transcript SMN-AS1 Regulates SMN Expression and Is a Novel Therapeutic Target for Spinal Muscular Atrophy.

Authors:  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
Journal:  Neuron       Date:  2016-12-22       Impact factor: 17.173

6.  Bifunctional antisense oligonucleotides provide a trans-acting splicing enhancer that stimulates SMN2 gene expression in patient fibroblasts.

Authors:  Leigh A Skordis; Matthew G Dunckley; Baigong Yue; Ian C Eperon; Francesco Muntoni
Journal:  Proc Natl Acad Sci U S A       Date:  2003-03-17       Impact factor: 11.205

7.  Reduced levels of survival motor neuron protein leads to aberrant motoneuron growth in a Xenopus model of muscular atrophy.

Authors:  Qods Ymlahi-Ouazzani; Odile J Bronchain; Elodie Paillard; Chantal Ballagny; Albert Chesneau; Aurélie Jadaud; André Mazabraud; Nicolas Pollet
Journal:  Neurogenetics       Date:  2009-06-11       Impact factor: 2.660

8.  Cell-specific survival motor neuron gene expression during human development of the central nervous system: implications for the pathogenesis of spinal muscular atrophy.

Authors:  E F Tizzano; C Cabot; M Baiget
Journal:  Am J Pathol       Date:  1998-08       Impact factor: 4.307

9.  Selective loss of alpha motor neurons with sparing of gamma motor neurons and spinal cord cholinergic neurons in a mouse model of spinal muscular atrophy.

Authors:  Rachael A Powis; Thomas H Gillingwater
Journal:  J Anat       Date:  2015-11-17       Impact factor: 2.610

Review 10.  Spinal muscular atrophy: journeying from bench to bedside.

Authors:  Tomoyuki Awano; Jeong-Ki Kim; Umrao R Monani
Journal:  Neurotherapeutics       Date:  2014-10       Impact factor: 7.620

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.