Literature DB >> 21179609

Therapeutic developments in spinal muscular atrophy.

Douglas M Sproule1, Petra Kaufmann.   

Abstract

Spinal muscular atrophy (SMA), a potentially devastating disease marked by progressive weakness and muscle atrophy resulting from the dysfunction and loss of motor neurons of the spinal cord, has emerged in recent years as an attractive target for therapeutic intervention. Caused by a homozygous mutation to the Survival of Motor Neurons 1 (SMN1) gene on chromosome 5q, the severity of the clinical phenotype in SMA is modulated by the function of a related protein, Survival of Motor Neurons 2 (SMN2). SMN2 predominantly produces an unstable SMN transcript lacking exon 7; only about 10% of the transcription product produces a full-length, functional SMN protein. Several therapeutic strategies have targeted this gene with the goal of producing increased full-length SMN transcript, thereby modifying the underlying mechanism. Drugs that have increased SMN2 function, in vitro, are now explored for potential therapeutic benefit in this disease. Alternative approaches, including neuroprotective, muscle anabolic, gene and cell replacement strategies, also hold promise. The recent advances in preclinical research and the development of a wider range of animal models for SMA continue to provide cautious optimism that effective treatments for SMA will eventually emerge.

Entities:  

Keywords:  clinical trials; gene therapy; spinal muscular atrophy; treatment

Year:  2010        PMID: 21179609      PMCID: PMC3002649          DOI: 10.1177/1756285610369026

Source DB:  PubMed          Journal:  Ther Adv Neurol Disord        ISSN: 1756-2856            Impact factor:   6.570


  105 in total

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Authors:  Q Liu; G Dreyfuss
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3.  Delivery of recombinant follistatin lessens disease severity in a mouse model of spinal muscular atrophy.

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5.  Doxycycline-controlled splicing modulation by regulated antisense U7 snRNA expression cassettes.

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7.  De novo and inherited deletions of the 5q13 region in spinal muscular atrophies.

Authors:  J Melki; S Lefebvre; L Burglen; P Burlet; O Clermont; P Millasseau; S Reboullet; B Bénichou; M Zeviani; D Le Paslier
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8.  Epidemiology of neuromuscular disorders in the under-20 population in Bologna Province, Italy.

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

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10.  A negatively acting bifunctional RNA increases survival motor neuron both in vitro and in vivo.

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Journal:  Hum Gene Ther       Date:  2008-11       Impact factor: 4.793

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Journal:  ACS Chem Neurosci       Date:  2012-01-18       Impact factor: 4.418

4.  Perceptions of equine-assisted activities and therapies by parents and children with spinal muscular atrophy.

Authors:  Danielle Lemke; Erin Rothwell; Tara M Newcomb; Kathryn J Swoboda
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5.  Severe burn increased skeletal muscle loss in mdx mutant mice.

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6.  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
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7.  Expression of Muscle-Specific MiRNA 206 in the Progression of Disease in a Murine SMA Model.

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8.  A New Method for Measuring Bell-Shaped Chest Induced by Impaired Ribcage Muscles in Spinal Muscular Atrophy Children.

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  8 in total

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