McLear J A, Lebrecht D, Messer A, Wolfgang W J
Division of Genetic Disorders, Wadsworth Center, New York State Department of Health, 120 New Scotland Ave., Albany, NY 12208, USA.
FASEB J. 2008 Jun;22(6):2003-11. doi: 10.1096/fj.07-099689. Epub 2008 Jan 16.
Intracellular antibodies (intrabodies) and the chaperone, heat shock protein 70 (Hsp70), have each shown potential as therapeutics for neurodegenerative diseases in vitro and in vivo. Investigating combinational therapy in an established Drosophila model of Huntington's disease (HD), we show that Hsp70 and intrabody actually affect different aspects of the disease. Overexpression of human Hsp70 resulted in improved survival of HD flies to eclosion and prolonged adult life compared with intrabody treatment alone. An additive effect on adult survival was observed when the two therapies were combined. Intrabody was more successful at suppressing neurodegeneration in photoreceptors than was Hsp70. Furthermore, Hsp70 treatment alone did not block aggregation of mutant huntingtin, a process slowed by intrabody. Expression of each is restricted to the nervous system, which implies different neuronal populations respond distinctly to these treatments. Importantly, a role for endogenous Hsp70 in suppression of mutant huntingtin pathology was confirmed by a separate set of genetic studies in which HD flies deficient for Hsp70 showed significantly increased pathology. We conclude that a combinational approach of intrabody with enhanced Hsp70 expression is beneficial in addressing multiple pathologies associated with HD and has potential application for other neurodegenerative disorders.
细胞内抗体(胞内抗体)和伴侣蛋白热休克蛋白70(Hsp70)在体外和体内均已显示出作为神经退行性疾病治疗药物的潜力。在已建立的亨廷顿舞蹈病(HD)果蝇模型中研究联合疗法时,我们发现Hsp70和胞内抗体实际上影响该疾病的不同方面。与单独使用胞内抗体治疗相比,人Hsp70的过表达导致HD果蝇存活至羽化的情况得到改善,并且成虫寿命延长。当两种疗法联合使用时,观察到对成虫存活有相加作用。胞内抗体在抑制光感受器中的神经退行性变方面比Hsp70更成功。此外,单独使用Hsp70治疗不能阻止突变型亨廷顿蛋白的聚集,而胞内抗体可减缓这一过程。每种蛋白的表达都局限于神经系统,这意味着不同的神经元群体对这些治疗的反应明显不同。重要的是,通过另一组基因研究证实了内源性Hsp70在抑制突变型亨廷顿蛋白病理方面的作用,在这些研究中,缺乏Hsp70的HD果蝇显示出明显增加的病理变化。我们得出结论,胞内抗体与增强的Hsp70表达相结合的方法有利于解决与HD相关的多种病理问题,并对其他神经退行性疾病具有潜在的应用价值。