Wang Jiou, Xu Guilian, Borchelt David R
Department of Neuroscience, Johns Hopkins School of Medicine, Baltimore, Maryland 21205, USA.
Neurobiol Dis. 2002 Mar;9(2):139-48. doi: 10.1006/nbdi.2001.0471.
Mutations in the cytosolic enzyme, superoxide dismutase 1, have been identified as the cause of motor neuron disease in a subset of cases of familial amyotrophic lateral sclerosis. It has been postulated that the injurious property of mutant enzyme resides in its propensity to aggregate or its propensity to catalyze deleterious, copper-mediated, chemistries. Aggregates of SOD1 have been identified, histologically, in neurons and astroglia of the spinal cords of SOD1-linked FALS patients and in transgenic mice that express these mutant proteins. In the present study, we have employed a technique used in detecting and quantifying aggregates of mutant huntingtin (cellulose acetate filtration) to examine the molecular characteristics of mutant SOD1 in three previously characterized transgenic mouse models of FALS. We show that the brains and spinal cords of these mice accumulate mutant SOD1 complexes that can be trapped by cellulose acetate filtration. The relative abundance of these structures increases dramatically with age. Although expressed to the same level in nonnervous tissues, mutant SOD1 was not found in high molecular weight structures. We conclude that some aspect of the biology of neural tissues (in a setting of declining motor neuron function) predisposes to the accumulation of high molecular weight complexes of mutant SOD1.
胞质酶超氧化物歧化酶1(SOD1)的突变已被确定为家族性肌萎缩侧索硬化症部分病例中运动神经元疾病的病因。据推测,突变酶的有害特性在于其聚集倾向或催化有害的、铜介导的化学反应的倾向。在组织学上,已在与SOD1相关的家族性肌萎缩侧索硬化症(FALS)患者脊髓的神经元和星形胶质细胞以及表达这些突变蛋白的转基因小鼠中鉴定出SOD1聚集体。在本研究中,我们采用了一种用于检测和定量突变亨廷顿蛋白聚集体的技术(醋酸纤维素过滤法),来研究三种先前已表征的FALS转基因小鼠模型中突变SOD1的分子特征。我们发现,这些小鼠的大脑和脊髓中积累了可被醋酸纤维素过滤捕获的突变SOD1复合物。这些结构的相对丰度随年龄显著增加。尽管突变SOD1在非神经组织中的表达水平相同,但在高分子量结构中未发现。我们得出结论,在运动神经元功能下降的情况下,神经组织生物学的某些方面易导致突变SOD1高分子量复合物的积累。