Department of Neuroscience and Physiology, New York University School of Medicine, New York, NY 10016, United States of America.
Department of Cell Biology, New York University School of Medicine, New York, NY 10016, United States of America.
Neurobiol Dis. 2020 Apr;137:104770. doi: 10.1016/j.nbd.2020.104770. Epub 2020 Jan 23.
We have derived single-chain variable fragments (scFv) from tau antibody hybridomas and previously shown their promise as imaging diagnostic agents. Here, we examined the therapeutic potential of anti-tau scFv in transgenic Drosophila models that express in neurons wild-type (WT) human tau (htau) or the human tauopathy mutation R406W. scFv expressing flies were crossed with the tauopathy flies and analyzed. Overall, the survival curves differed significantly (p < .0001). Control flies not expressing htau survived the longest, whereas R406W expressing flies had the shortest lifespan, which was greatly prolonged by co-expressing the anti-tau scFv (p < .0001). Likewise, htau WT expressing flies had a moderately short lifespan, which was prolonged by co-expressing the anti-tau scFv (p < .01). In addition, the htau expression impaired wing expansion after eclosion (p < .0001), and caused progressive abdomen expansion (p < .0001). These features were more severe in htau R406W flies than in htau WT flies. Importantly, both phenotypes were prevented by co-expression of the anti-tau scFv (p < .01-0.0001). Lastly, brain analyses revealed scFv-mediated tau clearance (p < .05-0.01), and its prevention of tau-mediated neurotoxicity (p < .05-0.001). In summary, these findings support the therapeutic potential of an anti-tau scFv, including as gene therapies, and the use of Drosophila models for such screening.
我们从 tau 抗体杂交瘤中提取了单链可变片段 (scFv),并已证明其作为成像诊断试剂具有潜力。在此,我们在表达神经元中野生型 (WT) 人 tau (htau) 或人 tau 病变突变 R406W 的转基因果蝇模型中研究了抗 tau scFv 的治疗潜力。表达 scFv 的果蝇与 tau 病变果蝇交配并进行分析。总的来说,生存曲线差异显著 (p <.0001)。不表达 htau 的对照果蝇存活时间最长,而表达 R406W 的果蝇寿命最短,通过共表达抗 tau scFv 大大延长 (p <.0001)。同样,表达 htau WT 的果蝇寿命适中较短,通过共表达抗 tau scFv 延长 (p <.01)。此外,htau 表达在羽化后损害翅膀扩张 (p <.0001),并导致腹部进行性扩张 (p <.0001)。在 htau R406W 果蝇中比在 htau WT 果蝇中更严重。重要的是,共表达抗 tau scFv 可预防这两种表型 (p <.01-0.0001)。最后,大脑分析显示 scFv 介导 tau 清除 (p <.05-0.01),并预防 tau 介导的神经毒性 (p <.05-0.001)。总之,这些发现支持抗 tau scFv 的治疗潜力,包括作为基因治疗,以及使用果蝇模型进行此类筛选。