生物标记证据表明,5xFAD 小鼠的早期视力和杆状能量相关病理生理学可从极低剂量的二甲基亚砜中获益。

IF 6.2 2区 医学 Q1 NEUROSCIENCES
Bruce A Berkowitz, Anuhya Paruchuri, Josh Stanek, Mura Abdul-Nabi, Robert H Podolsky, Abner Heredia Bustos, Karen Lins Childers, Geoffrey G Murphy, Katherine Stangis, Robin Roberts
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引用次数: 0

摘要

在这里,我们测试了烟酰胺核苷酸转氢酶(Nnt)无效的 5xFAD 小鼠是否也会出现表明病理生理学的早期视觉和 OCT 杆状能量相关生物标记物,以及这些生物标记物是否可以通过药物治疗。研究对象是四个月大的野生型或 5xFAD C57BL/6 亚系的 Nnt 基因无效(B6J)或完整的 Nnt 基因(B6NTac)小鼠,以及用 R-carvedilol + 车辆或仅用车辆(0.01% DMSO)治疗一个月的 5xFAD B6J 小鼠。研究人员测量了小鼠的对比敏感度(CS)、外部限界膜-视网膜色素上皮(ELM-RPE)厚度(低pH值触发的水分去除的代表)、ELM正后方高反射带的轮廓形状(即感光器内线粒体配置的单位长宽比[MCP/AR])以及视网膜板层厚度。两个野生型亚系都表现出相似的视觉表现指数和暗诱发的ELM-RPE收缩。与 B6J 小鼠不同的是,B6NTac MCP/AR 没有光-暗变化,这与线粒体效率相对较高是一致的。5xFAD B6J 小鼠的 CS 值低于 WT 值,而 5xFAD B6NTac 小鼠的 CS 值则低于 WT 值。光适应 5xFAD 亚系均表现出异常的 ELM-RPE 收缩和高于 WT 的 MCP/AR 收缩。视网膜内层和视网膜外层较薄。用 R-carvedilol + DMSO 或仅用 DMSO 治疗 5xFAD B6J 小鼠可纠正 CS 和 ELM-RPE 收缩,但不能纠正 MCP/AR 超常收缩或薄层化。这些结果为前驱光感受器线粒体功能障碍/氧化应激/氧化损伤提供了生物标志物证据,这与视觉表现以及 Nnt 基因的存在无关。5xFAD 小鼠的这种病理生理学是可以药物治疗的。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Biomarker evidence of early vision and rod energy-linked pathophysiology benefits from very low dose DMSO in 5xFAD mice.

Here, we test whether early visual and OCT rod energy-linked biomarkers indicating pathophysiology in nicotinamide nucleotide transhydrogenase (Nnt)-null 5xFAD mice also occur in Nnt-intact 5xFAD mice and whether these biomarkers can be pharmacologically treated. Four-month-old wild-type or 5xFAD C57BL/6 substrains with either a null (B6J) Nnt or intact Nnt gene (B6NTac) and 5xFAD B6J mice treated for one month with either R-carvedilol + vehicle or only vehicle (0.01% DMSO) were studied. The contrast sensitivity (CS), external limiting membrane-retinal pigment epithelium (ELM-RPE) thickness (a proxy for low pH-triggered water removal), profile shape of the hyperreflective band just posterior to the ELM (i.e., the mitochondrial configuration within photoreceptors per aspect ratio [MCP/AR]), and retinal laminar thickness were measured. Both wild-type substrains showed similar visual performance indices and dark-evoked ELM-RPE contraction. The lack of a light-dark change in B6NTac MCP/AR, unlike in B6J mice, is consistent with relatively greater mitochondrial efficiency. 5xFAD B6J mice, but not 5xFAD B6NTac mice, showed lower-than-WT CS. Light-adapted 5xFAD substrains both showed abnormal ELM-RPE contraction and greater-than-WT MCP/AR contraction. The inner retina and superior outer retina were thinner. Treating 5xFAD B6J mice with R-carvedilol + DMSO or DMSO alone corrected CS and ELM-RPE contraction but not supernormal MCP/AR contraction or laminar thinning. These results provide biomarker evidence for prodromal photoreceptor mitochondrial dysfunction/oxidative stress/oxidative damage, which is unrelated to visual performance, as well as the presence of the Nnt gene. This pathophysiology is druggable in 5xFAD mice.

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来源期刊
Acta Neuropathologica Communications
Acta Neuropathologica Communications Medicine-Pathology and Forensic Medicine
CiteScore
11.20
自引率
2.80%
发文量
162
审稿时长
8 weeks
期刊介绍: "Acta Neuropathologica Communications (ANC)" is a peer-reviewed journal that specializes in the rapid publication of research articles focused on the mechanisms underlying neurological diseases. The journal emphasizes the use of molecular, cellular, and morphological techniques applied to experimental or human tissues to investigate the pathogenesis of neurological disorders. ANC is committed to a fast-track publication process, aiming to publish accepted manuscripts within two months of submission. This expedited timeline is designed to ensure that the latest findings in neuroscience and pathology are disseminated quickly to the scientific community, fostering rapid advancements in the field of neurology and neuroscience. The journal's focus on cutting-edge research and its swift publication schedule make it a valuable resource for researchers, clinicians, and other professionals interested in the study and treatment of neurological conditions.
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