Lopes Carla, Ribeiro Márcio, Duarte Ana I, Humbert Sandrine, Saudou Frederic, Pereira de Almeida Luís, Hayden Michael, Rego A Cristina
CNC-Center for Neuroscience and Cell Biology and Faculty of Medicine, University of Coimbra, Coimbra, Portugal.
Mol Neurobiol. 2014 Jun;49(3):1126-42. doi: 10.1007/s12035-013-8585-5. Epub 2013 Dec 18.
Huntington's disease (HD) is an autosomal dominant disease caused by an expansion of CAG repeats in the gene encoding for huntingtin. Brain metabolic dysfunction and altered Akt signaling pathways have been associated with disease progression. Nevertheless, conflicting results persist regarding the role of insulin-like growth factor-1 (IGF-1)/Akt pathway in HD. While high plasma levels of IGF-1 correlated with cognitive decline in HD patients, other data showed protective effects of IGF-1 in HD striatal neurons and R6/2 mice. Thus, in the present study, we investigated motor phenotype, peripheral and central metabolic profile, and striatal and cortical signaling pathways in YAC128 mice subjected to intranasal administration of recombinant human IGF-1 (rhIGF-1) for 2 weeks, in order to promote IGF-1 delivery to the brain. We show that IGF-1 supplementation enhances IGF-1 cortical levels and improves motor activity and both peripheral and central metabolic abnormalities in YAC128 mice. Moreover, decreased Akt activation in HD mice brain was ameliorated following IGF-1 administration. Upregulation of Akt following rhIGF-1 treatment occurred concomitantly with increased phosphorylation of mutant huntingtin on Ser421. These data suggest that intranasal administration of rhIGF-1 ameliorates HD-associated glucose metabolic brain abnormalities and mice phenotype.
亨廷顿舞蹈症(HD)是一种常染色体显性疾病,由编码亨廷顿蛋白的基因中CAG重复序列的扩增引起。脑代谢功能障碍和Akt信号通路改变与疾病进展有关。然而,关于胰岛素样生长因子-1(IGF-1)/Akt通路在HD中的作用,仍存在相互矛盾的结果。虽然HD患者血浆中高水平的IGF-1与认知能力下降相关,但其他数据显示IGF-1对HD纹状体神经元和R6/2小鼠具有保护作用。因此,在本研究中,我们对经鼻内给予重组人IGF-1(rhIGF-1)2周的YAC128小鼠的运动表型、外周和中枢代谢概况以及纹状体和皮质信号通路进行了研究,以促进IGF-1向脑内传递。我们发现,补充IGF-1可提高YAC128小鼠皮质中的IGF-1水平,并改善其运动活性以及外周和中枢代谢异常。此外,给予IGF-1后,HD小鼠脑中降低的Akt激活得到改善。rhIGF-1治疗后Akt的上调与突变型亨廷顿蛋白Ser421位点磷酸化增加同时发生。这些数据表明,经鼻内给予rhIGF-1可改善与HD相关的脑葡萄糖代谢异常和小鼠表型。