Literature DB >> 26681261

Is spinal muscular atrophy a disease of the motor neurons only: pathogenesis and therapeutic implications?

Chiara Simone1, Agnese Ramirez1, Monica Bucchia1, Paola Rinchetti1, Hardy Rideout2, Dimitra Papadimitriou2, Diane B Re3,4, Stefania Corti5.   

Abstract

Spinal muscular atrophy (SMA) is a genetic neurological disease that causes infant mortality; no effective therapies are currently available. SMA is due to homozygous mutations and/or deletions in the survival motor neuron 1 gene and subsequent reduction of the SMN protein, leading to the death of motor neurons. However, there is increasing evidence that in addition to motor neurons, other cell types are contributing to SMA pathology. In this review, we will discuss the involvement of non-motor neuronal cells, located both inside and outside the central nervous system, in disease onset and progression. Even if SMN restoration in motor neurons is needed, it has been shown that optimal phenotypic amelioration in animal models of SMA requires a more widespread SMN correction. It has been demonstrated that non-motor neuronal cells are also involved in disease pathogenesis and could have important therapeutic implications. For these reasons it will be crucial to take this evidence into account for the clinical translation of the novel therapeutic approaches.

Entities:  

Keywords:  Central nervous system; Pathogenesis; Spinal muscular atrophy; Therapy

Mesh:

Year:  2015        PMID: 26681261      PMCID: PMC4756905          DOI: 10.1007/s00018-015-2106-9

Source DB:  PubMed          Journal:  Cell Mol Life Sci        ISSN: 1420-682X            Impact factor:   9.261


  143 in total

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Review 4.  Glia-to-neuron signaling and the neuroendocrine control of female puberty.

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5.  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

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7.  SP600125, a new JNK inhibitor, protects dopaminergic neurons in the MPTP model of Parkinson's disease.

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8.  Sphingosine 1-phosphate induces cell contraction via calcium-independent/Rho-dependent pathways in undifferentiated skeletal muscle cells.

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Journal:  J Cell Physiol       Date:  2004-01       Impact factor: 6.384

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Journal:  J Neurophysiol       Date:  2012-11-07       Impact factor: 2.714

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  27 in total

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2.  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
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Review 3.  Comparison of high-dose intracisterna magna and lumbar puncture intrathecal delivery of AAV9 in mice to treat neuropathies.

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Review 4.  Muscle-Bone Interactions in Pediatric Bone Diseases.

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5.  Downregulation of Survivin contributes to cell-cycle arrest during postnatal cardiac development in a severe spinal muscular atrophy mouse model.

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Journal:  Hum Mol Genet       Date:  2018-02-01       Impact factor: 6.150

6.  Gestational Age-Dependent Increase of Survival Motor Neuron Protein in Umbilical Cord-Derived Mesenchymal Stem Cells.

Authors:  Sota Iwatani; Nur Imma Fatimah Harahap; Dian Kesumapramudya Nurputra; Shinya Tairaku; Akemi Shono; Daisuke Kurokawa; Keiji Yamana; Khin Kyae Mon Thwin; Makiko Yoshida; Masami Mizobuchi; Tsubasa Koda; Kazumichi Fujioka; Mariko Taniguchi-Ikeda; Hideto Yamada; Ichiro Morioka; Kazumoto Iijima; Hisahide Nishio; Noriyuki Nishimura
Journal:  Front Pediatr       Date:  2017-09-05       Impact factor: 3.418

Review 7.  RNA-binding proteins in neurodegeneration: mechanisms in aggregate.

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9.  Functional Abnormalities of Cerebellum and Motor Cortex in Spinal Muscular Atrophy Mice.

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Review 10.  In Search of a Cure: The Development of Therapeutics to Alter the Progression of Spinal Muscular Atrophy.

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