Literature DB >> 33562482

In Search of a Cure: The Development of Therapeutics to Alter the Progression of Spinal Muscular Atrophy.

Kristine S Ojala1, Emily J Reedich2,3, Christine J DiDonato2,3, Stephen D Meriney1.   

Abstract

Until the recent development of disease-modifying therapeutics, spinal muscular atrophy (SMA) was considered a devastating neuromuscular disease with a poor prognosis for most affected individuals. Symptoms generally present during early childhood and manifest as muscle weakness and progressive paralysis, severely compromising the affected individual's quality of life, independence, and lifespan. SMA is most commonly caused by the inheritance of homozygously deleted SMN1 alleles with retention of one or more copies of a paralog gene, SMN2, which inversely correlates with disease severity. The recent advent and use of genetically targeted therapies have transformed SMA into a prototype for monogenic disease treatment in the era of genetic medicine. Many SMA-affected individuals receiving these therapies achieve traditionally unobtainable motor milestones and survival rates as medicines drastically alter the natural progression of this disease. This review discusses historical SMA progression and underlying disease mechanisms, highlights advances made in therapeutic research, clinical trials, and FDA-approved medicines, and discusses possible second-generation and complementary medicines as well as optimal temporal intervention windows in order to optimize motor function and improve quality of life for all SMA-affected individuals.

Entities:  

Keywords:  motoneuron disease; neuromuscular disease; spinal muscular atrophy; therapeutics

Year:  2021        PMID: 33562482      PMCID: PMC7915832          DOI: 10.3390/brainsci11020194

Source DB:  PubMed          Journal:  Brain Sci        ISSN: 2076-3425


  307 in total

1.  Correlation between severity and SMN protein level in spinal muscular atrophy.

Authors:  S Lefebvre; P Burlet; Q Liu; S Bertrandy; O Clermont; A Munnich; G Dreyfuss; J Melki
Journal:  Nat Genet       Date:  1997-07       Impact factor: 38.330

2.  Pharmacologically induced mouse model of adult spinal muscular atrophy to evaluate effectiveness of therapeutics after disease onset.

Authors:  Zhihua Feng; Karen K Y Ling; Xin Zhao; Chunyi Zhou; Gary Karp; Ellen M Welch; Nikolai Naryshkin; Hasane Ratni; Karen S Chen; Friedrich Metzger; Sergey Paushkin; Marla Weetall; Chien-Ping Ko
Journal:  Hum Mol Genet       Date:  2016-01-11       Impact factor: 6.150

3.  Loganin possesses neuroprotective properties, restores SMN protein and activates protein synthesis positive regulator Akt/mTOR in experimental models of spinal muscular atrophy.

Authors:  Yu-Ting Tseng; Cheng-Sheng Chen; Yuh-Jyh Jong; Fang-Rong Chang; Yi-Ching Lo
Journal:  Pharmacol Res       Date:  2016-05-27       Impact factor: 7.658

Review 4.  Diagnosis and management of spinal muscular atrophy: Part 1: Recommendations for diagnosis, rehabilitation, orthopedic and nutritional care.

Authors:  Eugenio Mercuri; Richard S Finkel; Francesco Muntoni; Brunhilde Wirth; Jacqueline Montes; Marion Main; Elena S Mazzone; Michael Vitale; Brian Snyder; Susana Quijano-Roy; Enrico Bertini; Rebecca Hurst Davis; Oscar H Meyer; Anita K Simonds; Mary K Schroth; Robert J Graham; Janbernd Kirschner; Susan T Iannaccone; Thomas O Crawford; Simon Woods; Ying Qian; Thomas Sejersen
Journal:  Neuromuscul Disord       Date:  2017-11-23       Impact factor: 4.296

5.  A single administration of morpholino antisense oligomer rescues spinal muscular atrophy in mouse.

Authors:  Paul N Porensky; Chalermchai Mitrpant; Vicki L McGovern; Adam K Bevan; Kevin D Foust; Brain K Kaspar; Stephen D Wilton; Arthur H M Burghes
Journal:  Hum Mol Genet       Date:  2011-12-20       Impact factor: 6.150

6.  Association between Ag1-CA alleles and severity of autosomal recessive proximal spinal muscular atrophy.

Authors:  C J DiDonato; K Morgan; J D Carpten; P Fuerst; S E Ingraham; G Prescott; J D McPherson; B Wirth; K Zerres; O Hurko
Journal:  Am J Hum Genet       Date:  1994-12       Impact factor: 11.025

7.  Spectrum of neuropathophysiology in spinal muscular atrophy type I.

Authors:  Brian N Harding; Shingo Kariya; Umrao R Monani; Wendy K Chung; Maryjane Benton; Sabrina W Yum; Gihan Tennekoon; Richard S Finkel
Journal:  J Neuropathol Exp Neurol       Date:  2015-01       Impact factor: 3.685

8.  Regular exercise prolongs survival in a type 2 spinal muscular atrophy model mouse.

Authors:  Clément Grondard; Olivier Biondi; Anne-Sophie Armand; Sylvie Lécolle; Bruno Della Gaspera; Claude Pariset; Hung Li; Claude-Louis Gallien; Pierre-Paul Vidal; Christophe Chanoine; Frédéric Charbonnier
Journal:  J Neurosci       Date:  2005-08-17       Impact factor: 6.167

9.  Deletions of the survival motor neuron gene in unaffected siblings of patients with spinal muscular atrophy.

Authors:  J M Cobben; G van der Steege; P Grootscholten; M de Visser; H Scheffer; C H Buys
Journal:  Am J Hum Genet       Date:  1995-10       Impact factor: 11.025

10.  Unaffected patients with a homozygous absence of the SMN1 gene.

Authors:  Maria Jedrzejowska; Janina Borkowska; Janusz Zimowski; Anna Kostera-Pruszczyk; Michał Milewski; Marta Jurek; Danuta Sielska; Ewa Kostyk; Walenty Nyka; Jacek Zaremba; Irena Hausmanowa-Petrusewicz
Journal:  Eur J Hum Genet       Date:  2008-03-12       Impact factor: 4.246

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  2 in total

Review 1.  Therapy development for spinal muscular atrophy: perspectives for muscular dystrophies and neurodegenerative disorders.

Authors:  Sibylle Jablonka; Luisa Hennlein; Michael Sendtner
Journal:  Neurol Res Pract       Date:  2022-01-04

Review 2.  Ethical Issues in Care and Treatment of Neuronal Ceroid Lipofuscinoses (NCL)-A Personal View.

Authors:  Alfried Kohlschütter
Journal:  Front Neurol       Date:  2021-06-25       Impact factor: 4.003

  2 in total

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