Du Yali, Wang David, Hu Qinglin, Lai Zhizhen, Yang Chen, He Huiwen, Wang Shuqing, Zhang Huijuan, Chen Peng, Li Zepeng, Chen Miao, Han Bing
Department of Hematology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.
Medical Zhongcheng Limited Company, Hangzhou, China.
J Cell Mol Med. 2024 Dec;28(23):e70295. doi: 10.1111/jcmm.70295.
PIGA mutation cannot fully explain the proliferative advantage of abnormal clones and thrombosis tendency in paroxysmal nocturnal haemoglobinuria (PNH), and additional genes may play a role, justifying further investigation. CD59+ and CD59- peripheral blood mononuclear cells from six PNH patients were sorted and subjected to whole-exon sequencing (WES) and whole-transcriptome sequencing respectively. Six age- and sex-matched healthy volunteers were enrolled as controls. Genes related to proliferation, immunity and thrombosis were selected by gene ontology (GO) analysis. The selected gene mutant alleles were then identified in the WES results for 40 patients and verified by the Sanger method in another 40 PNH patients. CD59+ and CD59- peripheral blood mononuclear cells from seven patients were sorted, and the RNA and protein expression levels of target genes were assessed via quantitative real-time PCR (RT-qPCR), flow cytometry and western blotting. The final selected genes were then related to clinical features to analyse. T-cell activation-related genes were upregulated, whereas platelet degranulation, coagulation, haemostasis, leukocyte proliferation and platelet activation-related genes were downregulated in both CD59+ and CD59- cells. The mRNA or protein expression levels of SELP, FLT1, NRP1 and vWF were either different from those in healthy controls or different between CD59+ and CD59- cells. Moreover, platelet aggregation was greater in patients with mutations in these genes than in patients without such mutations. Except for PIGA, other genes may be involved in the proliferation and coagulopathy that occur in PNH patients.
磷脂酰肌醇聚糖A(PIGA)突变不能完全解释阵发性夜间血红蛋白尿(PNH)中异常克隆的增殖优势和血栓形成倾向,可能还有其他基因发挥作用,因此有必要进一步研究。对6例PNH患者的CD59 +和CD59 -外周血单个核细胞进行分选,分别进行全外显子组测序(WES)和全转录组测序。选取6例年龄和性别匹配的健康志愿者作为对照。通过基因本体论(GO)分析筛选出与增殖、免疫和血栓形成相关的基因。然后在40例患者的WES结果中鉴定所选基因的突变等位基因,并通过桑格法在另外40例PNH患者中进行验证。对7例患者的CD59 +和CD59 -外周血单个核细胞进行分选,通过定量实时PCR(RT-qPCR)、流式细胞术和蛋白质印迹法评估靶基因的RNA和蛋白质表达水平。然后将最终选定的基因与临床特征相关联进行分析。在CD59 +和CD59 -细胞中,T细胞活化相关基因上调,而血小板脱颗粒、凝血、止血、白细胞增殖和血小板活化相关基因下调。SELP、FLT1、NRP1和vWF的mRNA或蛋白质表达水平与健康对照不同,或在CD59 +和CD59 -细胞之间存在差异。此外,这些基因发生突变的患者血小板聚集程度高于未发生此类突变的患者。除PIGA外,其他基因可能参与了PNH患者的增殖和凝血病过程。