Literature DB >> 19584893

Spinal muscular atrophy: why do low levels of survival motor neuron protein make motor neurons sick?

Arthur H M Burghes1, Christine E Beattie.   

Abstract

Many neurogenetic disorders are caused by the mutation of ubiquitously expressed genes. One such disorder, spinal muscular atrophy, is caused by loss or mutation of the survival motor neuron1 gene (SMN1), leading to reduced SMN protein levels and a selective dysfunction of motor neurons. SMN, together with partner proteins, functions in the assembly of small nuclear ribonucleoproteins (snRNPs), which are important for pre-mRNA splicing. It has also been suggested that SMN might function in the assembly of other ribonucleoprotein complexes. Two hypotheses have been proposed to explain the molecular dysfunction that gives rise to spinal muscular atrophy (SMA) and its specificity to a particular group of neurons. The first hypothesis states that the loss of SMN's well-known function in snRNP assembly causes an alteration in the splicing of a specific gene (or genes). The second hypothesis proposes that SMN is crucial for the transport of mRNA in neurons and that disruption of this function results in SMA.

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Year:  2009        PMID: 19584893      PMCID: PMC2853768          DOI: 10.1038/nrn2670

Source DB:  PubMed          Journal:  Nat Rev Neurosci        ISSN: 1471-003X            Impact factor:   34.870


  168 in total

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Authors:  U R Monani; J D McPherson; A H Burghes
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2.  Spliceosomal U snRNP core assembly: Sm proteins assemble onto an Sm site RNA nonanucleotide in a specific and thermodynamically stable manner.

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Review 5.  Biogenesis of small nuclear RNPs.

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10.  Intragenic telSMN mutations: frequency, distribution, evidence of a founder effect, and modification of the spinal muscular atrophy phenotype by cenSMN copy number.

Authors:  D W Parsons; P E McAndrew; S T Iannaccone; J R Mendell; A H Burghes; T W Prior
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