Literature DB >> 29422644

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

Ewout J N Groen1,2, Kevin Talbot3, Thomas H Gillingwater1,2.   

Abstract

Spinal muscular atrophy (SMA) is a devastating motor neuron disease that predominantly affects children and represents the most common cause of hereditary infant mortality. The condition results from deleterious variants in SMN1, which lead to depletion of the survival motor neuron protein (SMN). Now, 20 years after the discovery of this genetic defect, a major milestone in SMA and motor neuron disease research has been reached with the approval of the first disease-modifying therapy for SMA by US and European authorities - the antisense oligonucleotide nusinersen. At the same time, promising data from early-stage clinical trials of SMN1 gene therapy have indicated that additional therapeutic options are likely to emerge for patients with SMA in the near future. However, the approval of nusinersen has generated a number of immediate and substantial medical, ethical and financial implications that have the potential to resonate beyond the specific treatment of SMA. Here, we provide an overview of the rapidly evolving therapeutic landscape for SMA, highlighting current achievements and future opportunities. We also discuss how these developments are providing important lessons for the emerging second generation of combinatorial ('SMN-plus') therapies that are likely to be required to generate robust treatments that are effective across a patient's lifespan.

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Year:  2018        PMID: 29422644     DOI: 10.1038/nrneurol.2018.4

Source DB:  PubMed          Journal:  Nat Rev Neurol        ISSN: 1759-4758            Impact factor:   42.937


  131 in total

1.  Dawn of a new therapeutic era for spinal muscular atrophy.

Authors:  Thomas H Gillingwater
Journal:  Lancet       Date:  2016-12-07       Impact factor: 79.321

2.  Association of motor milestones, SMN2 copy and outcome in spinal muscular atrophy types 0-4.

Authors:  Renske I Wadman; Marloes Stam; Marleen Gijzen; Henny H Lemmink; Irina N Snoeck; Camiel A Wijngaarde; Kees P J Braun; Marja A G C Schoenmakers; Leonard H van den Berg; Dennis Dooijes; W-Ludo van der Pol
Journal:  J Neurol Neurosurg Psychiatry       Date:  2017-01-20       Impact factor: 10.154

3.  Safety and efficacy of olesoxime in patients with type 2 or non-ambulatory type 3 spinal muscular atrophy: a randomised, double-blind, placebo-controlled phase 2 trial.

Authors:  Enrico Bertini; Eric Dessaud; Eugenio Mercuri; Francesco Muntoni; Janbernd Kirschner; Carol Reid; Anna Lusakowska; Giacomo P Comi; Jean-Marie Cuisset; Jean-Louis Abitbol; Bruno Scherrer; Patricia Sanwald Ducray; Jeppe Buchbjerg; Eduardo Vianna; W Ludo van der Pol; Carole Vuillerot; Thomas Blaettler; Paulo Fontoura
Journal:  Lancet Neurol       Date:  2017-04-28       Impact factor: 44.182

4.  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

5.  Newborn blood spot screening test using multiplexed real-time PCR to simultaneously screen for spinal muscular atrophy and severe combined immunodeficiency.

Authors:  Jennifer L Taylor; Francis K Lee; Golriz Khadem Yazdanpanah; John F Staropoli; Mei Liu; John P Carulli; Chao Sun; Steven F Dobrowolski; W Harry Hannon; Robert F Vogt
Journal:  Clin Chem       Date:  2014-12-11       Impact factor: 8.327

6.  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

7.  ALS-causative mutations in FUS/TLS confer gain and loss of function by altered association with SMN and U1-snRNP.

Authors:  Shuying Sun; Shuo-Chien Ling; Jinsong Qiu; Claudio P Albuquerque; Yu Zhou; Seiya Tokunaga; Hairi Li; Haiyan Qiu; Anh Bui; Gene W Yeo; Eric J Huang; Kevin Eggan; Huilin Zhou; Xiang-Dong Fu; Clotilde Lagier-Tourenne; Don W Cleveland
Journal:  Nat Commun       Date:  2015-01-27       Impact factor: 14.919

8.  Reduced sensory synaptic excitation impairs motor neuron function via Kv2.1 in spinal muscular atrophy.

Authors:  Emily V Fletcher; Christian M Simon; John G Pagiazitis; Joshua I Chalif; Aleksandra Vukojicic; Estelle Drobac; Xiaojian Wang; George Z Mentis
Journal:  Nat Neurosci       Date:  2017-05-15       Impact factor: 24.884

9.  Content validity and clinical meaningfulness of the HFMSE in spinal muscular atrophy.

Authors:  Maria C Pera; Giorgia Coratti; Nicola Forcina; Elena S Mazzone; Mariacristina Scoto; Jacqueline Montes; Amy Pasternak; Anna Mayhew; Sonia Messina; Maria Sframeli; Marion Main; Robert Muni Lofra; Tina Duong; Danielle Ramsey; Sally Dunaway; Rachel Salazar; Lavinia Fanelli; Matthew Civitello; Roberto de Sanctis; Laura Antonaci; Leonardo Lapenta; Simona Lucibello; Marika Pane; John Day; Basil T Darras; Darryl C De Vivo; Francesco Muntoni; Richard Finkel; Eugenio Mercuri
Journal:  BMC Neurol       Date:  2017-02-23       Impact factor: 2.474

10.  SMN and coilin negatively regulate dyskerin association with telomerase RNA.

Authors:  Aaron R Poole; Michael D Hebert
Journal:  Biol Open       Date:  2016-06-15       Impact factor: 2.422

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  57 in total

Review 1.  Nusinersen: A Review in 5q Spinal Muscular Atrophy.

Authors:  Sheridan M Hoy
Journal:  CNS Drugs       Date:  2018-07       Impact factor: 5.749

Review 2.  Two breakthrough gene-targeted treatments for spinal muscular atrophy: challenges remain.

Authors:  Charlotte J Sumner; Thomas O Crawford
Journal:  J Clin Invest       Date:  2018-07-09       Impact factor: 14.808

3.  Lamin A/C dysregulation contributes to cardiac pathology in a mouse model of severe spinal muscular atrophy.

Authors:  Darija Šoltić; Hannah K Shorrock; Hazel Allardyce; Emma L Wilson; Ian Holt; Silvia A Synowsky; Sally L Shirran; Simon H Parson; Thomas H Gillingwater; Heidi R Fuller
Journal:  Hum Mol Genet       Date:  2019-11-01       Impact factor: 6.150

4.  Physiological and druggable skipping of immunoglobulin variable exons in plasma cells.

Authors:  Mohamad Omar Ashi; Nivine Srour; Jean-Marie Lambert; Anne Marchalot; Ophélie Martin; Sandrine Le Noir; Eric Pinaud; Maria Victoria Ayala; Christophe Sirac; Jérôme Saulière; Jérôme Moreaux; Michel Cogné; Laurent Delpy
Journal:  Cell Mol Immunol       Date:  2018-08-20       Impact factor: 11.530

5.  Automated screening of C. elegans neurodegeneration mutants enabled by microfluidics and image analysis algorithms.

Authors:  Ivan de Carlos Cáceres; Daniel A Porto; Ivan Gallotta; Pamela Santonicola; Josue Rodríguez-Cordero; Elia Di Schiavi; Hang Lu
Journal:  Integr Biol (Camb)       Date:  2018-09-17       Impact factor: 2.192

Review 6.  Disruption of RNA Metabolism in Neurological Diseases and Emerging Therapeutic Interventions.

Authors:  Julia K Nussbacher; Ricardos Tabet; Gene W Yeo; Clotilde Lagier-Tourenne
Journal:  Neuron       Date:  2019-04-17       Impact factor: 17.173

Review 7.  Restoring Protein Expression in Neuromuscular Conditions: A Review Assessing the Current State of Exon Skipping/Inclusion and Gene Therapies for Duchenne Muscular Dystrophy and Spinal Muscular Atrophy.

Authors:  Omar Sheikh; Toshifumi Yokota
Journal:  BioDrugs       Date:  2021-06-07       Impact factor: 5.807

Review 8.  Therapies for rare diseases: therapeutic modalities, progress and challenges ahead.

Authors:  Erik Tambuyzer; Benjamin Vandendriessche; Christopher P Austin; Philip J Brooks; Kristina Larsson; Katherine I Miller Needleman; James Valentine; Kay Davies; Stephen C Groft; Robert Preti; Tudor I Oprea; Marco Prunotto
Journal:  Nat Rev Drug Discov       Date:  2019-12-13       Impact factor: 84.694

Review 9.  RNA-binding proteins in neurological development and disease.

Authors:  Shavanie Prashad; Pallavi P Gopal
Journal:  RNA Biol       Date:  2020-08-30       Impact factor: 4.652

10.  Serum creatinine is a biomarker of progressive denervation in spinal muscular atrophy.

Authors:  Christiano R R Alves; Ren Zhang; Alec J Johnstone; Reid Garner; Pann H Nwe; Jennifer J Siranosian; Kathryn J Swoboda
Journal:  Neurology       Date:  2019-12-27       Impact factor: 9.910

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