Baida Gleb, Popko Brian, Wollmann Robert L, Stavrou Spyridon, Lin Wensheng, Tretiakova Maria, Krausz Thomas N, Roos Raymond P
Department of Neurology, University of Chicago Pritzker School of Medicine, Chicago, IL 60637, USA.
J Virol. 2008 Jun;82(12):5879-86. doi: 10.1128/JVI.02432-07. Epub 2008 Apr 9.
The DA strain of Theiler's murine encephalomyelitis virus (TMEV) causes a persistent central nervous system (CNS) infection of mice with a restricted virus gene expression and induces an inflammatory demyelinating disease that is thought to be immune mediated and a model of multiple sclerosis (MS). The relative contribution of virus vis-à-vis the immune system in the pathogenesis of DA-induced white matter disease remains unclear, as is also true in MS. To clarify the pathogenesis of DA-induced demyelination, we used Cre/loxP technology to generate a transgenic mouse that has tamoxifen (Tm)-inducible expression of a subgenomic segment of DA RNA in oligodendrocytes and Schwann cells. Tm-treated young transgenic mice developed progressive weakness leading to death, with abnormalities of oligodendrocytes and Schwann cells and demyelination, but without inflammation, demonstrating that DA virus can play a direct pathogenic role in demyelination. Tm treatment of mice at a later age resulted in milder disease, with evidence of peripheral nerve remyelination and focal fur depigmentation; surviving weak mice had persistent expression of the recombined transgene in the CNS, suggesting that the DA subgenomic segment can cause cellular dysfunction but not death, possibly similar to the situation seen during DA virus persistence. These studies demonstrate that DA RNA or a DA protein(s) is toxic to myelin-synthesizing cells. This Cre/loxP transgenic system allows for spatially and temporally controlled expression of the viral transgene and is valuable for clarifying nonimmune (and immune) mechanisms of demyelination induced by TMEV as well as other viruses.
泰勒氏鼠脑脊髓炎病毒(TMEV)的DA毒株可引起小鼠中枢神经系统(CNS)的持续性感染,病毒基因表达受限,并诱发一种炎性脱髓鞘疾病,该疾病被认为是免疫介导的,是多发性硬化症(MS)的一种模型。在DA诱导的白质疾病发病机制中,病毒相对于免疫系统的相对作用仍不清楚,MS的情况也是如此。为了阐明DA诱导脱髓鞘的发病机制,我们使用Cre/loxP技术构建了一种转基因小鼠,该小鼠在少突胶质细胞和雪旺细胞中具有他莫昔芬(Tm)诱导的DA RNA亚基因组片段表达。经Tm处理的年轻转基因小鼠出现进行性虚弱直至死亡,伴有少突胶质细胞和雪旺细胞异常及脱髓鞘,但无炎症,这表明DA病毒可在脱髓鞘中发挥直接致病作用。在较晚年龄对小鼠进行Tm处理导致疾病较轻,有周围神经重新髓鞘化和局部皮毛色素脱失的证据;存活的虚弱小鼠在中枢神经系统中持续表达重组转基因,这表明DA亚基因组片段可导致细胞功能障碍但不会导致死亡,这可能与DA病毒持续存在期间的情况类似。这些研究表明,DA RNA或DA蛋白对髓鞘合成细胞有毒性。这种Cre/loxP转基因系统允许病毒转基因在空间和时间上受到控制的表达,对于阐明TMEV以及其他病毒诱导的脱髓鞘的非免疫(和免疫)机制很有价值。