Burghes Arthur H M, Beattie Christine E
Department of Molecular and Cellular Biochemistry, The Ohio State University, Columbus, Ohio 43210, USA.
Nat Rev Neurosci. 2009 Aug;10(8):597-609. doi: 10.1038/nrn2670. Epub 2009 Jul 8.
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.
许多神经遗传性疾病是由普遍表达的基因突变引起的。其中一种疾病,脊髓性肌萎缩症,是由存活运动神经元1基因(SMN1)的缺失或突变导致的,从而导致SMN蛋白水平降低以及运动神经元的选择性功能障碍。SMN与伴侣蛋白一起,在小核核糖核蛋白(snRNP)的组装中发挥作用,而snRNP对前体mRNA剪接很重要。也有人提出,SMN可能在其他核糖核蛋白复合物的组装中发挥作用。已经提出了两种假说来解释导致脊髓性肌萎缩症(SMA)及其对特定神经元群体特异性的分子功能障碍。第一个假说是,SMN在snRNP组装中众所周知的功能丧失会导致特定基因(或多个基因)剪接的改变。第二个假说提出,SMN对神经元中mRNA的运输至关重要,并且这种功能的破坏会导致SMA。