Roher Alex E, Maarouf Chera L, Malek-Ahmadi Michael, Wilson Jeffrey, Kokjohn Tyler A, Daugs Ian D, Whiteside Charisse M, Kalback Walter M, Macias Mimi P, Jacobson Sandra A, Sabbagh Marwan N, Ghetti Bernardino, Beach Thomas G
The Longtine Center for Neurodegenerative Biochemistry, Banner Sun Health Research Institute Sun City, AZ 85351.
Am J Neurodegener Dis. 2013 Sep 18;2(3):187-207. eCollection 2013.
Alzheimer's disease (AD) dementia impacts all facets of higher order cognitive function and is characterized by the presence of distinctive pathological lesions in the gray matter (GM). The profound alterations in GM structure and function have fostered the view that AD impacts are primarily a consequence of GM damage. However, the white matter (WM) represents about 50% of the cerebrum and this area of the brain is substantially atrophied and profoundly abnormal in both sporadic AD (SAD) and familial AD (FAD). We examined the WM biochemistry by ELISA and Western blot analyses of key proteins in 10 FAD cases harboring mutations in the presenilin genes PSEN1 and PSEN2 as well as in 4 non-demented control (NDC) individuals and 4 subjects with SAD. The molecules examined were direct substrates of PSEN1 such as Notch-1 and amyloid precursor protein (APP). In addition, apolipoproteins, axonal transport molecules, cytoskeletal and structural proteins, neurotrophic factors and synaptic proteins were examined. PSEN-FAD subjects had, on average, higher amounts of WM amyloid-beta (Aβ) peptides compared to SAD, which may play a role in the devastating dysfunction of the brain. However, the PSEN-FAD mutations we examined did not produce uniform increases in the relative proportions of Aβ42 and exhibited substantial variability in total Aβ levels. These observations suggest that neurodegeneration and dementia do not depend solely on enhanced Aβ42 levels. Our data revealed additional complexities in PSEN-FAD individuals. Some direct substrates of γ-secretase, such as Notch, N-cadherin, Erb-B4 and APP, deviated substantially from the NDC group baseline for some, but not all, mutation types. Proteins that were not direct γ-secretase substrates, but play key structural and functional roles in the WM, likewise exhibited varied concentrations in the distinct PSEN mutation backgrounds. Detailing the diverse biochemical pathology spectrum of PSEN mutations may offer valuable insights into dementia progression and the design of effective therapeutic interventions for both SAD and FAD.
阿尔茨海默病(AD)痴呆会影响高阶认知功能的各个方面,其特征是灰质(GM)中存在独特的病理病变。GM结构和功能的深刻改变使人认为AD的影响主要是GM损伤的结果。然而,白质(WM)约占大脑的50%,在散发性AD(SAD)和家族性AD(FAD)中,大脑的这一区域均出现明显萎缩且严重异常。我们通过酶联免疫吸附测定(ELISA)和蛋白质免疫印迹分析,检测了10例携带早老素基因PSEN1和PSEN2突变的FAD病例、4例非痴呆对照(NDC)个体以及4例SAD患者大脑白质中的关键蛋白质。所检测的分子是PSEN1的直接底物,如Notch-1和淀粉样前体蛋白(APP)。此外,还检测了载脂蛋白、轴突运输分子、细胞骨架和结构蛋白、神经营养因子以及突触蛋白。与SAD相比,PSEN-FAD受试者大脑白质中淀粉样β蛋白(Aβ)肽的含量平均更高,这可能在大脑的严重功能障碍中起作用。然而,我们检测的PSEN-FAD突变并未使Aβ42的相对比例均匀增加,且总Aβ水平存在很大差异。这些观察结果表明,神经退行性变和痴呆并不完全取决于Aβ42水平的升高。我们的数据揭示了PSEN-FAD个体存在更多复杂性。γ-分泌酶的一些直接底物,如Notch、N-钙黏蛋白、表皮生长因子受体4(Erb-B4)和APP,对于某些(但不是所有)突变类型,其水平与NDC组基线有很大偏差。那些不是γ-分泌酶直接底物,但在WM中起关键结构和功能作用的蛋白质,在不同的PSEN突变背景下同样表现出不同的浓度。详细了解PSEN突变的多样生化病理谱,可能为痴呆的进展以及针对SAD和FAD的有效治疗干预设计提供有价值的见解。