Literature DB >> 23157239

SMN-inducing compounds for the treatment of spinal muscular atrophy.

Monique A Lorson1, Christian L Lorson.   

Abstract

Spinal muscular atrophy (SMA) is a leading genetic cause of infant mortality. A neurodegenerative disease, it is caused by loss of SMN1, although low, but essential, levels of SMN protein are produced by the nearly identical gene SMN2. While no effective treatment or therapy currently exists, a new wave of therapeutics has rapidly progressed from cell-based and preclinical animal models to the point where clinical trials have initiated for SMA-specific compounds. There are several reasons why SMA has moved relatively rapidly towards novel therapeutics, including: SMA is monogenic; the molecular understanding of SMN gene regulation has been building for nearly 20 years; and all SMA patients retain one or more copies of SMN2 that produces low levels of full-length, fully functional SMN protein. This review primarily focuses upon the biology behind the disease and examines SMN1- and SMN2-targeted therapeutics.

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Year:  2012        PMID: 23157239      PMCID: PMC3589915          DOI: 10.4155/fmc.12.131

Source DB:  PubMed          Journal:  Future Med Chem        ISSN: 1756-8919            Impact factor:   3.808


  181 in total

1.  Phenotypic heterogeneity of spinal muscular atrophy mapping to chromosome 5q11.2-13.3 (SMA 5q).

Authors:  T L Munsat; L Skerry; B Korf; B Pober; Y Schapira; G G Gascon; S M al-Rajeh; V Dubowitz; K Davies; L M Brzustowicz
Journal:  Neurology       Date:  1990-12       Impact factor: 9.910

2.  Partial restoration of cardio-vascular defects in a rescued severe model of spinal muscular atrophy.

Authors:  Monir Shababi; Javad Habibi; Lixin Ma; Jacqueline J Glascock; James R Sowers; Christian L Lorson
Journal:  J Mol Cell Cardiol       Date:  2012-01-17       Impact factor: 5.000

Review 3.  Spinal muscular atrophy--clinical and genetic correlations.

Authors:  K Zerres; B Wirth; S Rudnik-Schöneborn
Journal:  Neuromuscul Disord       Date:  1997-05       Impact factor: 4.296

4.  The splicing regulator Sam68 binds to a novel exonic splicing silencer and functions in SMN2 alternative splicing in spinal muscular atrophy.

Authors:  Simona Pedrotti; Pamela Bielli; Maria Paola Paronetto; Fabiola Ciccosanti; Gian Maria Fimia; Stefan Stamm; James L Manley; Claudio Sette
Journal:  EMBO J       Date:  2010-02-25       Impact factor: 11.598

5.  Identification of novel compounds that increase SMN protein levels using an improved SMN2 reporter cell assay.

Authors:  Jonathan J Cherry; Matthew C Evans; Jake Ni; Gregory D Cuny; Marcie A Glicksman; Elliot J Androphy
Journal:  J Biomol Screen       Date:  2012-01-10

6.  The survival of motor neuron (SMN) protein interacts with the mRNA-binding protein HuD and regulates localization of poly(A) mRNA in primary motor neuron axons.

Authors:  Claudia Fallini; Honglai Zhang; Yuehang Su; Vincenzo Silani; Robert H Singer; Wilfried Rossoll; Gary J Bassell
Journal:  J Neurosci       Date:  2011-03-09       Impact factor: 6.167

7.  Delivery of recombinant follistatin lessens disease severity in a mouse model of spinal muscular atrophy.

Authors:  Ferrill F Rose; Virginia B Mattis; Hansjörg Rindt; Christian L Lorson
Journal:  Hum Mol Genet       Date:  2008-12-12       Impact factor: 6.150

8.  Spinal muscular atrophy and a model for survival of motor neuron protein function in axonal ribonucleoprotein complexes.

Authors:  Wilfried Rossoll; Gary J Bassell
Journal:  Results Probl Cell Differ       Date:  2009

9.  Antisense masking of an hnRNP A1/A2 intronic splicing silencer corrects SMN2 splicing in transgenic mice.

Authors:  Yimin Hua; Timothy A Vickers; Hazeem L Okunola; C Frank Bennett; Adrian R Krainer
Journal:  Am J Hum Genet       Date:  2008-03-27       Impact factor: 11.025

10.  Trichostatin A increases SMN expression and survival in a mouse model of spinal muscular atrophy.

Authors:  Amy M Avila; Barrington G Burnett; Addis A Taye; Francesca Gabanella; Melanie A Knight; Parvana Hartenstein; Ziga Cizman; Nicholas A Di Prospero; Livio Pellizzoni; Kenneth H Fischbeck; Charlotte J Sumner
Journal:  J Clin Invest       Date:  2007-02-22       Impact factor: 14.808

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  16 in total

Review 1.  Assays for the identification and prioritization of drug candidates for spinal muscular atrophy.

Authors:  Jonathan J Cherry; Dione T Kobayashi; Maureen M Lynes; Nikolai N Naryshkin; Francesco Danilo Tiziano; Phillip G Zaworski; Lee L Rubin; Jill Jarecki
Journal:  Assay Drug Dev Technol       Date:  2014-08       Impact factor: 1.738

Review 2.  Advances in therapy for spinal muscular atrophy: promises and challenges.

Authors:  Ewout J N Groen; Kevin Talbot; Thomas H Gillingwater
Journal:  Nat Rev Neurol       Date:  2018-02-09       Impact factor: 42.937

3.  Morpholino antisense oligonucleotides targeting intronic repressor Element1 improve phenotype in SMA mouse models.

Authors:  Erkan Y Osman; Madeline R Miller; Kate L Robbins; Abby M Lombardi; Arleigh K Atkinson; Amanda J Brehm; Christian L Lorson
Journal:  Hum Mol Genet       Date:  2014-04-29       Impact factor: 6.150

4.  Short-term safety results from compassionate use of risdiplam in patients with spinal muscular atrophy in Germany.

Authors:  Andreas Hahn; René Günther; Albert Ludolph; Oliver Schwartz; Regina Trollmann; Patrick Weydt; Markus Weiler; Kathrin Neuland; Martin Sebastian Schwaderer; Tim Hagenacker
Journal:  Orphanet J Rare Dis       Date:  2022-07-19       Impact factor: 4.303

5.  Requirement of enhanced Survival Motoneuron protein imposed during neuromuscular junction maturation.

Authors:  Shingo Kariya; Teresa Obis; Caterina Garone; Turgay Akay; Fusako Sera; Shinichi Iwata; Shunichi Homma; Umrao R Monani
Journal:  J Clin Invest       Date:  2014-01-27       Impact factor: 14.808

Review 6.  Spinal muscular atrophy: a motor neuron disorder or a multi-organ disease?

Authors:  Monir Shababi; Christian L Lorson; Sabine S Rudnik-Schöneborn
Journal:  J Anat       Date:  2013-07-22       Impact factor: 2.610

7.  Defining the therapeutic window in a severe animal model of spinal muscular atrophy.

Authors:  Kate L Robbins; Jacqueline J Glascock; Erkan Y Osman; Madeline R Miller; Christian L Lorson
Journal:  Hum Mol Genet       Date:  2014-04-09       Impact factor: 6.150

8.  The DcpS inhibitor RG3039 improves survival, function and motor unit pathologies in two SMA mouse models.

Authors:  Rocky G Gogliotti; Herminio Cardona; Jasbir Singh; Sophie Bail; Carina Emery; Nancy Kuntz; Michael Jorgensen; Madel Durens; Bing Xia; Courtenay Barlow; Christopher R Heier; Heather L Plasterer; Vincent Jacques; Megerditch Kiledjian; Jill Jarecki; James Rusche; Christine J DiDonato
Journal:  Hum Mol Genet       Date:  2013-06-04       Impact factor: 6.150

Review 9.  Spinal muscular atrophy--recent therapeutic advances for an old challenge.

Authors:  Irene Faravelli; Monica Nizzardo; Giacomo P Comi; Stefania Corti
Journal:  Nat Rev Neurol       Date:  2015-05-19       Impact factor: 42.937

10.  Enhancement of SMN protein levels in a mouse model of spinal muscular atrophy using novel drug-like compounds.

Authors:  Jonathan J Cherry; Erkan Y Osman; Matthew C Evans; Sungwoon Choi; Xuechao Xing; Gregory D Cuny; Marcie A Glicksman; Christian L Lorson; Elliot J Androphy
Journal:  EMBO Mol Med       Date:  2013-06-05       Impact factor: 12.137

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