Strong Melissa K, Southwell Amber L, Yonan Jennifer M, Hayden Michael R, Macgregor Grant R, Thompson Leslie M, Steward Oswald
Department of Anatomy and Neurobiology, University of California Irvine, Irvine, CA, USA.
J Huntingtons Dis. 2012;1(2):221-41. doi: 10.3233/JHD-129005.
Mouse strain background can influence vulnerability to excitotoxic neuronal cell death and potentially modulate phenotypes in transgenic mouse models of human disease. Evidence supports a contribution of excitotoxicity to the selective death of medium spiny neurons in Huntington's disease (HD). Here, we assess whether strain differences in excitotoxic vulnerability influence striatal cell death in a knock-in mouse model of HD. Previous studies that evaluated resistance to excitotoxic lesions in several mouse models of HD had variable outcomes. In the present study, we directly compare one model on two different background strains to test the contribution of strain to excitotoxicity-mediated neurodegeneration. Mice of the FVB/N strain, which are highly vulnerable to excitotoxicity, become extremely resistant to quinolinic acid-induced striatal neurodegeneration with age, when carrying a huntingtin (Htt) allele expressing a HD transgene (CAG140). The resistance is much greater than the age-dependent resistance that has been previously reported in YAC128 mice. By 12 months of age, both heterozygous and homozygous FVB.CAG140 mice displayed virtually complete resistance to quinolinic acid-induced striatal neurodegeneration. A similar resistance develops in CAG140 mice on a C57BL/6N background although the effect size is smaller because C57BL/6N mice are already resistant due to genetic background. In a direct comparison with the YAC128 mice, FVB.CAG140 mice have greater resistance. FVB.CAG140 mice are also resistant to neurodegeneration following kainic acid-induced status epilepticus suggesting the existence of a common cellular mechanism that provides protection against multiple types of excitotoxic insult. These findings establish FVB.CAG140 mice as a useful model to investigate the cellular and molecular mechanisms that confer neuroprotection against excitotoxicity.
小鼠品系背景可影响对兴奋性毒性神经元细胞死亡的易感性,并可能调节人类疾病转基因小鼠模型中的表型。有证据支持兴奋性毒性在亨廷顿舞蹈病(HD)中对中等棘状神经元的选择性死亡起作用。在此,我们评估兴奋性毒性易感性的品系差异是否会影响HD基因敲入小鼠模型中的纹状体细胞死亡。先前评估几种HD小鼠模型对兴奋性毒性损伤抗性的研究结果各不相同。在本研究中,我们直接比较了两种不同背景品系上的一种模型,以测试品系对兴奋性毒性介导的神经变性的影响。FVB/N品系的小鼠对兴奋性毒性高度敏感,但当携带表达HD转基因(CAG140)的亨廷顿蛋白(Htt)等位基因时,随着年龄增长,它们对喹啉酸诱导的纹状体神经变性变得极具抗性。这种抗性远大于先前在YAC128小鼠中报道的年龄依赖性抗性。到12个月大时,杂合和纯合的FVB.CAG140小鼠对喹啉酸诱导的纹状体神经变性几乎完全具有抗性。C57BL/6N背景的CAG140小鼠也出现了类似的抗性,尽管效应大小较小,因为C57BL/6N小鼠由于遗传背景已经具有抗性。与YAC128小鼠直接比较时,FVB.CAG140小鼠具有更强的抗性。FVB.CAG140小鼠对 kainic 酸诱导的癫痫持续状态后的神经变性也具有抗性,这表明存在一种共同的细胞机制可提供针对多种类型兴奋性毒性损伤的保护。这些发现确立了FVB.CAG140小鼠作为研究赋予抗兴奋性毒性神经保护作用的细胞和分子机制的有用模型。