Department of Neurology, Center for Translational Neuromedicine, University of Rochester Medical Center, Rochester, NY, USA.
Neurotherapeutics. 2011 Oct;8(4):577-90. doi: 10.1007/s13311-011-0075-8.
Huntington's disease (HD) is an inherited, relentlessly progressive neurodegenerative disease with an invariably fatal outcome. HD is inherited in an autosomal dominant fashion, and is characterized pathologically by the loss of cortical and striatal neurons, and clinically by involuntary choreiform movements accompanied by progressive cognitive impairment and emotional lability. The disorder is caused by an expanded cystosine adenine guanine (CAG) tri-nucleotide repeat encoding polyglutamine (polyQ) in the first exon of the Huntingtin gene. There is a correlation between the number of CAG repeats and disease onset, such that in patients with CAG repeat lengths of 36 to 60, disease symptoms typically manifest after 35 years of age, whereas CAG repeat lengths >60 yield the more severe juvenile form of the disease. Even though mutant huntingtin is expressed throughout the brain, it is characterized by the selective degeneration of medium spiny neurons of the caudate and putamen, which heralds more widespread neuronal degeneration with disease progression. The mechanisms of cell dysfunction and death in HD have been the subjects of a number of studies, which have led to therapeutic strategies largely based on the amelioration of mutant huntingtin-related metabolic impairment and cellular toxicity. Each of these approaches has aimed to delay or stop the preferential degeneration of medium spiny neurons early in the disease course. Yet, in later stages of the disease, after cell death has become prominent, cell replacement therapy (whether by direct cell transplantation or by the mobilization of endogenous progenitors) may comprise a stronger potential avenue for therapy. In this review, we will consider recent progress in the transplantation of fetal striatal cells to the HD brain, as well as emerging alternative sources for human striatal progenitor cells. We will then consider the potential application of gene therapy toward the induction of striatal neurogenesis and neuronal recruitment, with an eye toward its potential therapeutic use in HD.
亨廷顿病(HD)是一种遗传性、进行性神经退行性疾病,最终结果无一例外地是致命的。HD 以常染色体显性方式遗传,病理学特征是皮质和纹状体神经元丧失,临床表现为不自主舞蹈样运动,伴有进行性认知障碍和情绪不稳定。该疾病是由亨廷顿基因第一外显子中编码多聚谷氨酰胺(polyQ)的扩展胞嘧啶腺嘌呤鸟嘌呤(CAG)三核苷酸重复序列引起的。CAG 重复次数与疾病发病之间存在相关性,即 CAG 重复长度为 36 到 60 的患者,疾病症状通常在 35 岁后出现,而 CAG 重复长度>60 则导致更严重的青少年型疾病。尽管突变型亨廷顿蛋白在整个大脑中都有表达,但它的特征是尾状核和壳核的中型棘突神经元选择性退化,这预示着随着疾病的进展,更广泛的神经元退化。HD 中细胞功能障碍和死亡的机制一直是许多研究的主题,这些研究导致了治疗策略主要基于改善突变亨廷顿相关代谢损伤和细胞毒性。这些方法中的每一种都旨在延迟或阻止疾病早期中型棘突神经元的优先退化。然而,在疾病的后期,细胞死亡变得明显后,细胞替代疗法(无论是通过直接细胞移植还是动员内源性祖细胞)可能成为更强大的治疗途径。在这篇综述中,我们将考虑将胎儿纹状体细胞移植到 HD 大脑中的最新进展,以及新兴的人类纹状体祖细胞替代来源。然后,我们将考虑基因治疗在诱导纹状体神经发生和神经元募集方面的潜在应用,着眼于其在 HD 中的潜在治疗用途。