Woodhouse Adele, Shepherd Claire E, Sokolova Anna, Carroll Victoria L, King Anna E, Halliday Glenda M, Dickson Tracey C, Vickers James C
Wicking Dementia Research and Education Centre and NeuroRepair Group, Menzies Research Institute, Private Bag 29, Hobart, TAS, 7001, Australia.
Acta Neuropathol. 2009 Jan;117(1):19-29. doi: 10.1007/s00401-008-0458-z. Epub 2008 Nov 18.
Most cases of Alzheimer's disease (AD) are sporadic in nature, although rarer familial AD (FAD) cases have provided important insights into major pathological disease mechanisms. Mutations in the presenilin 1 gene (PS1) are responsible for the majority of FAD cases, causing an earlier age of onset and more rapid progression to end-stage disease than seen in sporadic AD. We have investigated the cytoskeletal alterations in neuritic AD pathology in a cohort of FAD cases in comparison to sporadic AD and pathologically aged cases. Tau-immunoreactive neurofibrillary tangle (NFT) loads were similar between PS1 FAD and sporadic AD cases. Similarly, plaque loads, both beta-amyloid (Abeta) and thioflavine S, in PS1 FAD and sporadic AD cases were not significantly different; however, in pathologically aged cases, they were significantly lower than those in PS1 cases, but were not different from sporadic AD cases. The 'cotton wool' plaque characteristic of PS1 cases did not demonstrate a high density of dystrophic neurites compared to other Abeta plaque types, but did demonstrate a localised mass effect on the neuropil. Despite minimal differences in plaque and NFT loads, immunolabelling demonstrated clear phenotypic differences in the NFTs and dystrophic neurites in PS1 FAD cases. Presenilin-1 cases exhibited significantly (P < 0.05) more tau-positive NFTs that were immunolabelled by the antibody SMI312 (anti-phosphorylated NF protein and phosphorylated tau) than sporadic AD cases. Presenilin-1 cases also exhibited numerous ring-like NF-positive and elongated tau-labelled dystrophic neurites, whereas these dystrophic neurite types were only abundant at the very early (pathologically aged cases) or very late stages of sporadic AD progression, respectively. These differences in cytoskeletal pathology in PS1 cases suggest an accelerated rate of neuritic pathology development, potentially due to mutant PS1 influencing multiple pathogenic pathways.
大多数阿尔茨海默病(AD)病例本质上是散发性的,尽管较罕见的家族性AD(FAD)病例为主要病理疾病机制提供了重要见解。早老素1基因(PS1)的突变是大多数FAD病例的病因,与散发性AD相比,其发病年龄更早,疾病进展至终末期更快。我们调查了一组FAD病例与散发性AD病例及病理衰老病例相比,神经炎性AD病理中的细胞骨架改变。PS1 FAD病例和散发性AD病例之间tau免疫反应性神经原纤维缠结(NFT)负荷相似。同样,PS1 FAD病例和散发性AD病例中的斑块负荷,即β淀粉样蛋白(Aβ)和硫黄素S,没有显著差异;然而,在病理衰老病例中,它们显著低于PS1病例,但与散发性AD病例没有差异。与其他Aβ斑块类型相比,PS1病例的“棉絮状”斑块特征并未显示出高密度的营养不良性神经突,但确实显示出对神经毡的局部质量效应。尽管斑块和NFT负荷差异最小,但免疫标记显示PS1 FAD病例的NFT和营养不良性神经突存在明显的表型差异。早老素-1病例表现出显著(P < 0.05)更多的tau阳性NFT,这些NFT被抗体SMI312(抗磷酸化NF蛋白和磷酸化tau)免疫标记,比散发性AD病例更多。早老素-1病例还表现出许多环状NF阳性和细长的tau标记的营养不良性神经突,而这些营养不良性神经突类型分别仅在散发性AD进展的非常早期(病理衰老病例)或非常晚期丰富。PSI病例中细胞骨架病理的这些差异表明神经炎性病理发展速度加快,可能是由于突变的PS1影响了多种致病途径。