Literature DB >> 31973611

New treatments in spinal muscular atrophy: an overview of currently available data.

Sithara Ramdas1, Laurent Servais1,2.   

Abstract

Introduction: Spinal muscular atrophy (SMA) is one of the most common inherited neuromuscular disorders. It causes progressive muscle weakness and results in significant disability. Until recently, there were no drugs available for the treatment of SMA. Several phase 1-3 studies, including three double-blind randomized placebo-controlled studies have demonstrated the efficacy of disease-modifying approaches including gene replacement therapy, antisense oligonucleotides, and splicing modifiers.Areas covered: This article covers the publically available data on therapeutic strategies that address the underlying cause of SMA and clinical data available on approved treatments and drugs in the pipeline.Expert opinion: The newer therapeutic options in SMA have a good safety profile and deliver a therapeutic benefit in most patients. It is essential that the recommended standards of care are delivered along with the drugs for the best outcomes. No biomarkers to distinguish responders from non-responders are available; it is important that biomarkers be identified. Early treatment is essential for the maximum efficacy of the newly available treatments.

Entities:  

Keywords:  Spinal muscular atrophy; antisense oligonucleotide; branaplam; gene therapy; nusinersen; risdiplam; splicing modifiers

Year:  2020        PMID: 31973611     DOI: 10.1080/14656566.2019.1704732

Source DB:  PubMed          Journal:  Expert Opin Pharmacother        ISSN: 1465-6566            Impact factor:   3.889


  24 in total

1.  Calibration of Pathogenicity Due to Variant-Induced Leaky Splicing Defects by Using BRCA2 Exon 3 as a Model System.

Authors:  Hélène Tubeuf; Sandrine M Caputo; Teresa Sullivan; Julie Rondeaux; Sophie Krieger; Virginie Caux-Moncoutier; Julie Hauchard; Gaia Castelain; Alice Fiévet; Laëtitia Meulemans; Françoise Révillion; Mélanie Léoné; Nadia Boutry-Kryza; Capucine Delnatte; Marine Guillaud-Bataille; Linda Cleveland; Susan Reid; Eileen Southon; Omar Soukarieh; Aurélie Drouet; Daniela Di Giacomo; Myriam Vezain; Françoise Bonnet-Dorion; Violaine Bourdon; Hélène Larbre; Danièle Muller; Pascal Pujol; Fátima Vaz; Séverine Audebert-Bellanger; Chrystelle Colas; Laurence Venat-Bouvet; Angela R Solano; Dominique Stoppa-Lyonnet; Claude Houdayer; Thierry Frebourg; Pascaline Gaildrat; Shyam K Sharan; Alexandra Martins
Journal:  Cancer Res       Date:  2020-07-08       Impact factor: 12.701

Review 2.  The effect of scoliosis surgery on pulmonary function in spinal muscular atrophy patients: review of the literature and a meta-analysis.

Authors:  Maria Angeli; Kalliopi Alpantaki; Nikolaos Pandis; Christos Koutserimpas; Alexander Hadjipavlou
Journal:  Eur Spine J       Date:  2022-06-04       Impact factor: 2.721

3.  Onasemnogene abeparvovec for presymptomatic infants with two copies of SMN2 at risk for spinal muscular atrophy type 1: the Phase III SPR1NT trial.

Authors:  Kevin A Strauss; Michelle A Farrar; Francesco Muntoni; Kayoko Saito; Jerry R Mendell; Laurent Servais; Hugh J McMillan; Richard S Finkel; Kathryn J Swoboda; Jennifer M Kwon; Craig M Zaidman; Claudia A Chiriboga; Susan T Iannaccone; Jena M Krueger; Julie A Parsons; Perry B Shieh; Sarah Kavanagh; Sitra Tauscher-Wisniewski; Bryan E McGill; Thomas A Macek
Journal:  Nat Med       Date:  2022-06-17       Impact factor: 87.241

4.  Onasemnogene abeparvovec for presymptomatic infants with three copies of SMN2 at risk for spinal muscular atrophy: the Phase III SPR1NT trial.

Authors:  Kevin A Strauss; Michelle A Farrar; Francesco Muntoni; Kayoko Saito; Jerry R Mendell; Laurent Servais; Hugh J McMillan; Richard S Finkel; Kathryn J Swoboda; Jennifer M Kwon; Craig M Zaidman; Claudia A Chiriboga; Susan T Iannaccone; Jena M Krueger; Julie A Parsons; Perry B Shieh; Sarah Kavanagh; Melissa Wigderson; Sitra Tauscher-Wisniewski; Bryan E McGill; Thomas A Macek
Journal:  Nat Med       Date:  2022-06-17       Impact factor: 87.241

Review 5.  Current Status of Antisense Oligonucleotide-Based Therapy in Neuromuscular Disorders.

Authors:  Flavien Bizot; Adeline Vulin; Aurélie Goyenvalle
Journal:  Drugs       Date:  2020-09       Impact factor: 9.546

Review 6.  Drug Screening and Drug Repositioning as Promising Therapeutic Approaches for Spinal Muscular Atrophy Treatment.

Authors:  Giovanna Menduti; Daniela Maria Rasà; Serena Stanga; Marina Boido
Journal:  Front Pharmacol       Date:  2020-11-12       Impact factor: 5.810

7.  The authors reply: Letter on: "Myostatin inhibition in combination with antisense oligonucleotide therapy improves outcomes in spinal muscular atrophy" by Zhou et al.

Authors:  Haiyan Zhou; Francesco Muntoni
Journal:  J Cachexia Sarcopenia Muscle       Date:  2020-07-23       Impact factor: 12.910

Review 8.  Williams syndrome.

Authors:  Beth A Kozel; Boaz Barak; Chong Ae Kim; Carolyn B Mervis; Lucy R Osborne; Melanie Porter; Barbara R Pober
Journal:  Nat Rev Dis Primers       Date:  2021-06-17       Impact factor: 65.038

Review 9.  Spinal muscular atrophy care in the COVID-19 pandemic era.

Authors:  Aravindhan Veerapandiyan; Anne M Connolly; Richard S Finkel; Kapil Arya; Katherine D Mathews; Edward C Smith; Diana Castro; Russell J Butterfield; Julie A Parsons; Laurent Servais; Nancy Kuntz; Vamshi K Rao; John F Brandsema; Eugenio Mercuri; Emma Ciafaloni
Journal:  Muscle Nerve       Date:  2020-05-03       Impact factor: 3.852

10.  Molecular Crosstalk Between Non-SMN-Related and SMN-Related Spinal Muscular Atrophy.

Authors:  Darija Šoltić; Heidi R Fuller
Journal:  Neurosci Insights       Date:  2020-03-30
View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.