Hedlund Eva, Hefferan Michael P, Marsala Martin, Isacson Ole
Neuroregeneration Laboratory, Center for Neuroregeneration Research, McLean Hospital/Harvard Medical School, Belmont, MA 02478, USA.
Eur J Neurosci. 2007 Oct;26(7):1721-37. doi: 10.1111/j.1460-9568.2007.05780.x.
Amyotrophic lateral sclerosis (ALS), spinal bulbar muscular atrophy (or Kennedy's disease), spinal muscular atrophy and spinal muscular atrophy with respiratory distress 1 are neurodegenerative disorders mainly affecting motor neurons and which currently lack effective therapies. Recent studies in animal models as well as primary and embryonic stem cell models of ALS, utilizing over-expression of mutated forms of Cu/Zn superoxide dismutase 1, have shown that motor neuron degeneration in these models is in part a non cell-autonomous event and that by providing genetically non-compromised supporting cells such as microglia or growth factor-excreting cells, onset can be delayed and survival increased. Using models of acute motor neuron injury it has been shown that embryonic stem cell-derived motor neurons implanted into the spinal cord can innervate muscle targets and improve functional recovery. Thus, a rationale exists for the development of cell therapies in motor neuron diseases aimed at either protecting and/or replacing lost motor neurons, interneurons as well as non-neuronal cells. This review evaluates approaches used in animal models of motor neuron disorders and their therapeutic relevance.
肌萎缩侧索硬化症(ALS)、脊髓延髓肌肉萎缩症(或肯尼迪病)、脊髓性肌萎缩症以及伴有呼吸窘迫的脊髓性肌萎缩症1型均为主要影响运动神经元的神经退行性疾病,目前缺乏有效的治疗方法。最近在动物模型以及ALS的原代和胚胎干细胞模型中的研究,利用突变形式的铜/锌超氧化物歧化酶1的过表达,表明这些模型中的运动神经元变性部分是非细胞自主性事件,并且通过提供基因上未受损的支持细胞,如小胶质细胞或分泌生长因子的细胞,可以延迟疾病发作并提高生存率。使用急性运动神经元损伤模型已表明,植入脊髓的胚胎干细胞衍生的运动神经元可以支配肌肉靶点并改善功能恢复。因此,存在开发针对运动神经元疾病的细胞疗法的理论基础,旨在保护和/或替代丢失的运动神经元、中间神经元以及非神经元细胞。本综述评估了在运动神经元疾病动物模型中使用的方法及其治疗相关性。