Department of Pediatrics and Developmental Biology, Tokyo Medical and Dental University Graduate School of Medicine, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8519, Japan.
Int J Hematol. 2012 Mar;95(3):299-310. doi: 10.1007/s12185-012-1013-1. Epub 2012 Feb 5.
Wiskott-Aldrich syndrome (WAS) is an X-linked immunodeficiency disease affecting cell morphology and signal transduction in hematopoietic cells. The function of Wiskott-Aldrich syndrome protein (WASp) and its partners in protein interaction have been studied intensively in mice; however, detailed biochemical characterization of its signal transduction and assessment of its functional consequence in human WASp-deficient lymphocytes remain difficult. In this study, we generated Nalm-6 cells in which the WAS protein gene (WASP) was disrupted by homologous recombination-based gene targeting and a cell-permeable form of recombinant WASp for functional study. The WASP⁻/⁻ cells showed impaired adhesive capacity and polarization to plate-bound anti-CD47 mAb, anti-CD9 mAb, or to fibronectin. The defective morphological changes were accompanied by impaired intracellular signaling. In addition, the WASp-deficient cells displayed augmented apoptosis induced by CD24 cross-linking. A recombinant fusion protein composed of Hph-1 cell-permeable peptide and WASp prepared in Escherichia coli. Hph-1-WASp was efficiently transduced and expressed in WASP⁻/⁻ Nalm-6 cells in a dose-dependent manner. The wild-type WASp, but not the mutant restored adhesion capacity, spreading morphology, and cytoskeletal reorganization. Additionally, the recombinant protein was successfully transduced into normal lymphocytes. These findings suggest that gene-disrupted model cell lines and cell-permeable recombinant proteins may serve as important tools for the detailed analysis of intracellular molecules involved in PID.
威特综合征(Wiskott-Aldrich syndrome,WAS)是一种 X 连锁免疫缺陷病,影响造血细胞的细胞形态和信号转导。Wiskott-Aldrich 综合征蛋白(Wiskott-Aldrich syndrome protein,WASp)及其蛋白相互作用伙伴的功能已在小鼠中进行了深入研究;然而,其信号转导的详细生化特征及其在人 WASp 缺陷淋巴细胞中的功能后果评估仍然较为困难。在本研究中,我们通过同源重组基因靶向技术使 Nalm-6 细胞中的 WAS 蛋白基因(WASP)发生缺失,并制备了一种细胞可渗透的重组 WASp 形式,用于功能研究。WASP-/-细胞表现出黏附能力受损,对板结合的抗 CD47 mAb、抗 CD9 mAb 或纤连蛋白的极化能力受损。形态缺陷伴随着细胞内信号转导受损。此外,WASP 缺陷细胞在 CD24 交联诱导的凋亡中表现出增强。一种由 Hph-1 细胞可渗透肽和 WASp 组成的重组融合蛋白在大肠杆菌中制备。Hph-1-WASp 以剂量依赖的方式有效地转导并表达于 WASP-/-Nalm-6 细胞中。野生型 WASp,但不是突变体,恢复了黏附能力、铺展形态和细胞骨架重排。此外,该重组蛋白成功转导至正常淋巴细胞中。这些发现表明基因缺失模型细胞系和细胞可渗透的重组蛋白可能成为分析 PID 相关细胞内分子的重要工具。