Literature DB >> 11504946

Treatment of spinal muscular atrophy by sodium butyrate.

J G Chang1, H M Hsieh-Li, Y J Jong, N M Wang, C H Tsai, H Li.   

Abstract

Spinal muscular atrophy (SMA) is an autosomal recessive disease characterized by degeneration of the anterior horn cells of the spinal cord, leading to muscular paralysis with muscular atrophy. No effective treatment of this disorder is presently available. Studies of the correlation between disease severity and the amount of survival motor neuron (SMN) protein have shown an inverse relationship. We report that sodium butyrate effectively increases the amount of exon 7-containing SMN protein in SMA lymphoid cell lines by changing the alternative splicing pattern of exon 7 in the SMN2 gene. In vivo, sodium butyrate treatment of SMA-like mice resulted in increased expression of SMN protein in motor neurons of the spinal cord and resulted in significant improvement of SMA clinical symptoms. Oral administration of sodium butyrate to intercrosses of heterozygous pregnant knockout-transgenic SMA-like mice decreased the birth rate of severe types of SMA-like mice, and SMA symptoms were ameliorated for all three types of SMA-like mice. These results suggest that sodium butyrate may be an effective drug for the treatment of human SMA patients.

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Year:  2001        PMID: 11504946      PMCID: PMC55534          DOI: 10.1073/pnas.171105098

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  38 in total

1.  An exonic enhancer is required for inclusion of an essential exon in the SMA-determining gene SMN.

Authors:  C L Lorson; E J Androphy
Journal:  Hum Mol Genet       Date:  2000-01-22       Impact factor: 6.150

Review 2.  RNA processing and human disease.

Authors:  A V Philips; T A Cooper
Journal:  Cell Mol Life Sci       Date:  2000-02       Impact factor: 9.261

3.  Htra2-beta 1 stimulates an exonic splicing enhancer and can restore full-length SMN expression to survival motor neuron 2 (SMN2).

Authors:  Y Hofmann; C L Lorson; S Stamm; E J Androphy; B Wirth
Journal:  Proc Natl Acad Sci U S A       Date:  2000-08-15       Impact factor: 11.205

4.  The human centromeric survival motor neuron gene (SMN2) rescues embryonic lethality in Smn(-/-) mice and results in a mouse with spinal muscular atrophy.

Authors:  U R Monani; M Sendtner; D D Coovert; D W Parsons; C Andreassi; T T Le; S Jablonka; B Schrank; W Rossoll; W Rossol; T W Prior; G E Morris; A H Burghes
Journal:  Hum Mol Genet       Date:  2000-02-12       Impact factor: 6.150

5.  Sodium butyrate inhibits histone deacetylation in cultured cells.

Authors:  E P Candido; R Reeves; J R Davie
Journal:  Cell       Date:  1978-05       Impact factor: 41.582

6.  The effect of sodium butyrate on histone modification.

Authors:  L Sealy; R Chalkley
Journal:  Cell       Date:  1978-05       Impact factor: 41.582

7.  Analysis of the mRNA transcripts of the survival motor neuron (SMN) gene in the tissue of an SMA fetus and the peripheral blood mononuclear cells of normals, carriers and SMA patients.

Authors:  Y J Jong; J G Chang; S P Lin; T Y Yang; J C Wang; C P Chang; C C Lee; H Li; H M Hsieh-Li; C H Tsai
Journal:  J Neurol Sci       Date:  2000-02-15       Impact factor: 3.181

8.  Intragenic telSMN mutations: frequency, distribution, evidence of a founder effect, and modification of the spinal muscular atrophy phenotype by cenSMN copy number.

Authors:  D W Parsons; P E McAndrew; S T Iannaccone; J R Mendell; A H Burghes; T W Prior
Journal:  Am J Hum Genet       Date:  1998-12       Impact factor: 11.025

9.  Butyric acid, a potent inducer of erythroid differentiation in cultured erythroleukemic cells.

Authors:  A Leder; P Leder
Journal:  Cell       Date:  1975-07       Impact factor: 41.582

10.  Activation of a chicken embryonic globin gene in adult erythroid cells by 5-azacytidine and sodium butyrate.

Authors:  G D Ginder; M J Whitters; J K Pohlman
Journal:  Proc Natl Acad Sci U S A       Date:  1984-07       Impact factor: 11.205

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  129 in total

1.  Genetic adaptation controlled by methylations and acetylations at the nuclear and cytosolic levels: a hypothetical model.

Authors:  Maurice Israël
Journal:  Neurochem Res       Date:  2003-04       Impact factor: 3.996

2.  In vivo selection reveals combinatorial controls that define a critical exon in the spinal muscular atrophy genes.

Authors:  Natalia N Singh; Elliot J Androphy; Ravindra N Singh
Journal:  RNA       Date:  2004-08       Impact factor: 4.942

3.  Creating a pro-survival and anti-inflammatory phenotype by modulation of acetylation in models of hemorrhagic and septic shock.

Authors:  Yongqing Li; Hasan B Alam
Journal:  Adv Exp Med Biol       Date:  2012       Impact factor: 2.622

Review 4.  Spinal muscular atrophy: new and emerging insights from model mice.

Authors:  Gyu-Hwan Park; Shingo Kariya; Umrao R Monani
Journal:  Curr Neurol Neurosci Rep       Date:  2010-03       Impact factor: 5.081

Review 5.  Spinal muscular atrophy: an update on therapeutic progress.

Authors:  Joonbae Seo; Matthew D Howell; Natalia N Singh; Ravindra N Singh
Journal:  Biochim Biophys Acta       Date:  2013-08-27

Review 6.  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

Review 7.  Perspectives on clinical trials in spinal muscular atrophy.

Authors:  Kathryn J Swoboda; John T Kissel; Thomas O Crawford; Mark B Bromberg; Gyula Acsadi; Guy D'Anjou; Kristin J Krosschell; Sandra P Reyna; Mary K Schroth; Charles B Scott; Louise R Simard
Journal:  J Child Neurol       Date:  2007-08       Impact factor: 1.987

8.  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

Review 9.  Spinal muscular atrophy.

Authors:  Susan T Iannaccone; Stephen A Smith; Louise R Simard
Journal:  Curr Neurol Neurosci Rep       Date:  2004-01       Impact factor: 5.081

Review 10.  Multiple roles of HDAC inhibition in neurodegenerative conditions.

Authors:  De-Maw Chuang; Yan Leng; Zoya Marinova; Hyeon-Ju Kim; Chi-Tso Chiu
Journal:  Trends Neurosci       Date:  2009-09-21       Impact factor: 13.837

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