Alzheimer's Disease and Other Cognitive Disorders Unit, Neurology Service, Hospital Clínic de Barcelona, IDIBAPS, Institut de Neurociències, Universitat de Barcelona, 08036, Barcelona, Spain.
Statistics and Bioinformatics Unit, Vall d'Hebrón Institut de Recerca, Passeig Vall d'Hebrón, Barcelona, Spain.
Mol Neurobiol. 2022 Oct;59(10):6411-6428. doi: 10.1007/s12035-022-02969-2. Epub 2022 Aug 12.
Sporadic early-onset Alzheimer's disease (EOAD) and autosomal dominant Alzheimer's disease (ADAD) provide the opportunity to investigate the physiopathological mechanisms in the absence of aging, present in late-onset forms. Frontotemporal dementia (FTD) causes early-onset dementia associated to tau or TDP43 protein deposits. A 15% of FTD cases are caused by mutations in C9orf72, GRN, or MAPT genes. Lymphoblastoid cell lines (LCLs) have been proposed as an alternative to brain tissue for studying earlier phases of neurodegenerative diseases. The aim of this study is to investigate the expression profile in EOAD, ADAD, and sporadic and genetic FTD (sFTD and gFTD, respectively), using brain tissue and LCLs. Sixty subjects of the following groups were included: EOAD, ADAD, sFTD, gFTD, and controls. Gene expression was analyzed with Clariom D microarray (Affymetrix). Brain tissue pairwise comparisons revealed six common differentially expressed genes (DEG) for all the patients' groups compared with controls: RGS20, WIF1, HSPB1, EMP3, S100A11 and GFAP. Common up-regulated biological pathways were identified both in brain and LCLs (including inflammation and glial cell differentiation), while down-regulated pathways were detected mainly in brain tissue (including synaptic signaling, metabolism and mitochondrial dysfunction). CD163, ADAMTS9 and LIN7A gene expression disruption was validated by qPCR in brain tissue and NrCAM in LCLs in their respective group comparisons. In conclusion, our study highlights neuroinflammation, metabolism and synaptic signaling disturbances as common altered pathways in different AD and FTD forms. The use of LCLs might be appropriate for studying early immune system and inflammation, and some neural features in neurodegenerative dementias.
散发性早发性阿尔茨海默病(EOAD)和常染色体显性阿尔茨海默病(ADAD)为研究晚发性疾病中存在的与衰老无关的生理病理机制提供了机会。额颞叶痴呆(FTD)导致与tau 或 TDP43 蛋白沉积相关的早发性痴呆。15%的 FTD 病例由 C9orf72、GRN 或 MAPT 基因突变引起。淋巴母细胞系(LCL)已被提议作为脑组织的替代物,用于研究神经退行性疾病的早期阶段。本研究旨在使用脑组织和 LCL 研究 EOAD、ADAD 以及散发性和遗传性 FTD(分别为 sFTD 和 gFTD)的表达谱。纳入了以下 60 名受试者的组别:EOAD、ADAD、sFTD、gFTD 和对照组。使用 Clariom D 微阵列(Affymetrix)分析基因表达。脑组织两两比较显示,与对照组相比,所有患者组共有的六个差异表达基因(DEG):RGS20、WIF1、HSPB1、EMP3、S100A11 和 GFAP。在大脑和 LCL 中均鉴定到共同上调的生物学途径(包括炎症和神经胶质细胞分化),而下调的途径主要在脑组织中检测到(包括突触信号、代谢和线粒体功能障碍)。在大脑组织中通过 qPCR 验证了 CD163、ADAMTS9 和 LIN7A 基因表达的中断,在 LCL 中验证了 NrCAM 基因在各自的组比较中的表达中断。总之,本研究强调了神经炎症、代谢和突触信号紊乱是不同 AD 和 FTD 形式中共同改变的途径。LCL 的使用可能适合于研究神经退行性痴呆症中的早期免疫系统和炎症以及一些神经特征。