b细胞淋巴瘤/白血病11A (BCL11A)基因在β-地中海贫血治疗中的新作用

Nahil Hassan Mahmoud, Mei I Lai
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引用次数: 1

摘要

β-地中海贫血是一种遗传性疾病,导致β-珠蛋白基因表达减少或缺失。由于β-地中海贫血的高患病率以及除了输血和造血干细胞(HSC)移植之外缺乏可用的治疗方法,该疾病对临床和经济系统构成了相当大的负担。胎儿血红蛋白在β-地中海贫血中具有明显的改善作用,因为在β-地中海贫血中,γ-珠蛋白链通过与过量的α-珠蛋白链配对来替代β-珠蛋白链的减少,并减少镰状细胞病(SCD)中的镰状细胞病。BCL11A是胎儿血红蛋白的关键调节因子和抑制因子。BCL11A在成人红细胞和表达成人血红蛋白的细胞系中的下调导致胎儿血红蛋白水平显著升高。BCL11A红系增强子的破坏导致BCL11A基因在红系谱系中被破坏,而在成年红系细胞中γ-珠蛋白的表达增加。自体造血干细胞基因治疗是一种有吸引力的治疗选择,可以克服与异体移植相关的免疫并发症和供体可用性。利用基因组编辑技术,破坏BCL11A诱导造血干细胞中γ-珠蛋白表达已成为治疗β-地中海贫血的另一种方法。利用CRISPR-Cas9或碱基编辑器靶向γ-基因启动子上的+58 BCL11A红系增强子或BCL11A结合基序,成功地破坏了该基因和结合基序,随后HbF水平增加。本文概述了BCL11A在γ-珠蛋白基因沉默中的关键作用,并讨论了下调BCL11A作为改善β-地中海贫血手段的不同基因组编辑方法。
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The Novel Role of the B-Cell Lymphoma/Leukemia 11A (BCL11A) Gene in β-Thalassaemia Treatment.

β-thalassaemia is a genetic disorder resulting in a reduction or absence of β-globin gene expression. Due to the high prevalence of β-thalassaemia and the lack of available treatment other than blood transfusion and haematopoietic stem cell (HSC) transplantation, the disease represents a considerable burden to clinical and economic systems. Foetal haemoglobin has an appreciated ameliorating effect in β-haemoglobinopathy, as the γ-globin chain substitutes the β-globin chain reduction by pairing with the excess α-globin chain in β-thalassaemia and reduces sickling in sickle cell disease (SCD). BCL11A is a critical regulator and repressor of foetal haemoglobin. Downregulation of BCL11A in adult erythroblasts and cell lines expressing adult haemoglobin led to a significant increase in foetal haemoglobin levels. Disruption of BCL11A erythroid enhancer resulted in disruption of the BCL11A gene solely in the erythroid lineages and increased γ-globin expression in adult erythroid cells. Autologous haematopoietic stem cell gene therapy represents an attractive treatment option to overcome the immune complications and donor availability associated with allogeneic transplantation. Using genome editing technologies, the disruption of BCL11A to induce γ- globin expression in HSCs has emerged as an alternative approach to treat β-thalassaemia. Targeting the +58 BCL11A erythroid enhancer or BCL11A binding motif at the γ-gene promoter with CRISPR-Cas9 or base editors has successfully disrupted the gene and the binding motif with a subsequent increment in HbF levels. This review outlines the critical role of BCL11A in γ-globin gene silencing and discusses the different genome editing approaches to downregulate BCL11A as a means for ameliorating β-thalassaemia.

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来源期刊
Cardiovascular and Hematological Disorders - Drug Targets
Cardiovascular and Hematological Disorders - Drug Targets Medicine-Cardiology and Cardiovascular Medicine
CiteScore
1.90
自引率
0.00%
发文量
36
期刊介绍: Cardiovascular & Hematological Disorders - Drug Targets aims to cover all the latest and outstanding developments on the medicinal chemistry, pharmacology, molecular biology, genomics and biochemistry of contemporary molecular targets involved in cardiovascular and hematological disorders e.g. disease specific proteins, receptors, enzymes, genes. Each issue of the journal contains a series of timely in-depth reviews written by leaders in the field covering a range of current topics on drug targets involved in cardiovascular and hematological disorders. As the discovery, identification, characterization and validation of novel human drug targets for cardiovascular and hematological drug discovery continues to grow.
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