Simmons Danielle A, Casale Malcolm, Alcon Betzi, Pham Nha, Narayan Natasha, Lynch Gary
Department of Psychiatry and Human Behavior, University of California, Irvine, California 92697-4292, USA.
Glia. 2007 Aug 1;55(10):1074-84. doi: 10.1002/glia.20526.
Huntington's Disease (HD) is characterized primarily by neuropathological changes in the striatum, including loss of medium-spiny neurons, nuclear inclusions of the huntingtin protein, gliosis, and abnormally high iron levels. Information about how these conditions interact, or about the temporal order in which they appear, is lacking. This study investigated if, and when, iron-related changes occur in the R6/2 transgenic mouse model of HD and compared the results with those from HD patients. Relative to wild-type mice, R6/2 mice had increased immunostaining for ferritin, an iron storage protein, in the striatum beginning at 2-4 weeks postnatal and in cortex and hippocampus starting at 5-7 weeks. The ferritin staining was found primarily in microglia, and became more pronounced as the mice matured. Ferritin-labeled microglia in R6/2 mice appeared dystrophic in that they had thick, twisted processes with cytoplasmic breaks; some of these cells also contained the mutant huntingtin protein. Brains from HD patients (Vonsattel grades 0-4) also had increased numbers of ferritin-containing microglia, some of which were dystrophic. The cells were positive for Perl's stain, indicating that they contained abnormally high levels of iron. These results provide the first evidence that perturbations to iron metabolism in HD are predominately associated with microglia and occur early enough to be important contributors to HD progression.
亨廷顿舞蹈症(HD)的主要特征是纹状体出现神经病理学变化,包括中等棘状神经元丧失、亨廷顿蛋白的核内包涵体、胶质增生以及铁水平异常升高。目前尚缺乏关于这些情况如何相互作用或其出现的时间顺序的信息。本研究调查了HD的R6/2转基因小鼠模型中铁相关变化是否以及何时发生,并将结果与HD患者的结果进行比较。相对于野生型小鼠,R6/2小鼠从出生后2 - 4周开始纹状体中铁储存蛋白铁蛋白的免疫染色增加,从5 - 7周开始皮质和海马中的铁蛋白免疫染色增加。铁蛋白染色主要在小胶质细胞中发现,并且随着小鼠成熟而变得更加明显。R6/2小鼠中铁蛋白标记的小胶质细胞出现营养不良,其特征是具有粗大、扭曲的突起且伴有细胞质断裂;其中一些细胞还含有突变的亨廷顿蛋白。HD患者(冯·萨特尔分级0 - 4级)的大脑中含铁蛋白的小胶质细胞数量也增加,其中一些出现营养不良。这些细胞对佩尔氏染色呈阳性,表明它们含有异常高水平的铁。这些结果提供了首个证据,即HD中铁代谢紊乱主要与小胶质细胞相关,并且发生得足够早,足以成为HD进展的重要因素。