Literature DB >> 18485868

SMN deficiency causes tissue-specific perturbations in the repertoire of snRNAs and widespread defects in splicing.

Zhenxi Zhang1, Francesco Lotti, Kimberly Dittmar, Ihab Younis, Lili Wan, Mumtaz Kasim, Gideon Dreyfuss.   

Abstract

The survival of motor neurons (SMN) protein is essential for the biogenesis of small nuclear RNA (snRNA)-ribonucleoproteins (snRNPs), the major components of the pre-mRNA splicing machinery. Though it is ubiquitously expressed, SMN deficiency causes the motor neuron degenerative disease spinal muscular atrophy (SMA). We show here that SMN deficiency, similar to that which occurs in severe SMA, has unexpected cell type-specific effects on the repertoire of snRNAs and mRNAs. It alters the stoichiometry of snRNAs and causes widespread pre-mRNA splicing defects in numerous transcripts of diverse genes, preferentially those containing a large number of introns, in SMN-deficient mouse tissues. These findings reveal a key role for the SMN complex in RNA metabolism and in splicing regulation and indicate that SMA is a general splicing disease that is not restricted to motor neurons.

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Year:  2008        PMID: 18485868      PMCID: PMC2446403          DOI: 10.1016/j.cell.2008.03.031

Source DB:  PubMed          Journal:  Cell        ISSN: 0092-8674            Impact factor:   41.582


  62 in total

1.  Purification of native survival of motor neurons complexes and identification of Gemin6 as a novel component.

Authors:  Livio Pellizzoni; Jennifer Baccon; Juri Rappsilber; Matthias Mann; Gideon Dreyfuss
Journal:  J Biol Chem       Date:  2001-12-17       Impact factor: 5.157

2.  Identification and characterization of Gemin7, a novel component of the survival of motor neuron complex.

Authors:  Jennifer Baccon; Livio Pellizzoni; Juri Rappsilber; Matthias Mann; Gideon Dreyfuss
Journal:  J Biol Chem       Date:  2002-06-13       Impact factor: 5.157

3.  Newly synthesized small nuclear RNAs appear transiently in the cytoplasm.

Authors:  G W Zieve; R A Sauterer; R J Feeney
Journal:  J Mol Biol       Date:  1988-01-20       Impact factor: 5.469

Review 4.  hnRNP proteins and the biogenesis of mRNA.

Authors:  G Dreyfuss; M J Matunis; S Piñol-Roma; C G Burd
Journal:  Annu Rev Biochem       Date:  1993       Impact factor: 23.643

5.  Determinants of the interaction of the spinal muscular atrophy disease protein SMN with the dimethylarginine-modified box H/ACA small nucleolar ribonucleoprotein GAR1.

Authors:  Sarah E Whitehead; Kevin W Jones; Xing Zhang; Xiaodong Cheng; Rebecca M Terns; Michael P Terns
Journal:  J Biol Chem       Date:  2002-09-19       Impact factor: 5.157

6.  A novel function for SMN, the spinal muscular atrophy disease gene product, in pre-mRNA splicing.

Authors:  L Pellizzoni; N Kataoka; B Charroux; G Dreyfuss
Journal:  Cell       Date:  1998-11-25       Impact factor: 41.582

7.  Regulation of alternative splicing in vivo by overexpression of antagonistic splicing factors.

Authors:  J F Cáceres; S Stamm; D M Helfman; A R Krainer
Journal:  Science       Date:  1994-09-16       Impact factor: 47.728

8.  SMN, the spinal muscular atrophy protein, forms a pre-import snRNP complex with snurportin1 and importin beta.

Authors:  Usha Narayanan; Jason K Ospina; Mark R Frey; Michael D Hebert; A Gregory Matera
Journal:  Hum Mol Genet       Date:  2002-07-15       Impact factor: 6.150

9.  hnRNP I/PTB can antagonize the splicing repressor activity of SRp30c.

Authors:  Caroline Paradis; Philippe Cloutier; Lulzim Shkreta; Johanne Toutant; Klaus Klarskov; Benoit Chabot
Journal:  RNA       Date:  2007-06-04       Impact factor: 4.942

10.  Smn, the spinal muscular atrophy-determining gene product, modulates axon growth and localization of beta-actin mRNA in growth cones of motoneurons.

Authors:  Wilfried Rossoll; Sibylle Jablonka; Catia Andreassi; Ann-Kathrin Kröning; Kathrin Karle; Umrao R Monani; Michael Sendtner
Journal:  J Cell Biol       Date:  2003-11-17       Impact factor: 10.539

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  307 in total

1.  IPLEX administration improves motor neuron survival and ameliorates motor functions in a severe mouse model of spinal muscular atrophy.

Authors:  Michela Murdocca; Arianna Malgieri; Andrea Luchetti; Luciano Saieva; Gabriella Dobrowolny; Elvira de Leonibus; Antonio Filareto; Maria Chiara Quitadamo; Giuseppe Novelli; Antonio Musarò; Federica Sangiuolo
Journal:  Mol Med       Date:  2012-09-25       Impact factor: 6.354

2.  Post-transcriptional modification of spliceosomal RNAs is normal in SMN-deficient cells.

Authors:  Svetlana Deryusheva; Maria Choleza; Adrien Barbarossa; Joseph G Gall; Rémy Bordonné
Journal:  RNA       Date:  2011-11-28       Impact factor: 4.942

Review 3.  mRNA transcript diversity creates new opportunities for pharmacological intervention.

Authors:  Elizabeth S Barrie; Ryan M Smith; Jonathan C Sanford; Wolfgang Sadee
Journal:  Mol Pharmacol       Date:  2012-02-07       Impact factor: 4.436

Review 4.  RNA processing pathways in amyotrophic lateral sclerosis.

Authors:  Marka van Blitterswijk; John E Landers
Journal:  Neurogenetics       Date:  2010-03-27       Impact factor: 2.660

5.  The Prp19 complex and the Usp4Sart3 deubiquitinating enzyme control reversible ubiquitination at the spliceosome.

Authors:  Eun Joo Song; Shannon L Werner; Jakob Neubauer; Frank Stegmeier; Julie Aspden; Donald Rio; J Wade Harper; Stephen J Elledge; Marc W Kirschner; Michael Rape
Journal:  Genes Dev       Date:  2010-07-01       Impact factor: 11.361

Review 6.  Developments in RNA splicing and disease.

Authors:  Michael G Poulos; Ranjan Batra; Konstantinos Charizanis; Maurice S Swanson
Journal:  Cold Spring Harb Perspect Biol       Date:  2011-01-01       Impact factor: 10.005

Review 7.  The pathogenicity of splicing defects: mechanistic insights into pre-mRNA processing inform novel therapeutic approaches.

Authors:  Elisabeth Daguenet; Gwendal Dujardin; Juan Valcárcel
Journal:  EMBO Rep       Date:  2015-11-13       Impact factor: 8.807

8.  Multiple effects of curcumin on promoting expression of the exon 7-containing SMN2 transcript.

Authors:  Dairong Feng; Yi Cheng; Yan Meng; Liping Zou; Shangzhi Huang; Jiuyong Xie
Journal:  Genes Nutr       Date:  2015-09-19       Impact factor: 5.523

9.  The survival motor neuron gene smn-1 interacts with the U2AF large subunit gene uaf-1 to regulate Caenorhabditis elegans lifespan and motor functions.

Authors:  Xiaoyang Gao; Yanling Teng; Jintao Luo; Liange Huang; Min Li; Zhuohua Zhang; Yong-Chao Ma; Long Ma
Journal:  RNA Biol       Date:  2014       Impact factor: 4.652

10.  Genetic correction of human induced pluripotent stem cells from patients with spinal muscular atrophy.

Authors:  Stefania Corti; Monica Nizzardo; Chiara Simone; Marianna Falcone; Martina Nardini; Dario Ronchi; Chiara Donadoni; Sabrina Salani; Giulietta Riboldi; Francesca Magri; Giorgia Menozzi; Clara Bonaglia; Federica Rizzo; Nereo Bresolin; Giacomo P Comi
Journal:  Sci Transl Med       Date:  2012-12-19       Impact factor: 17.956

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