创建新的顺式调节HBD元件以重新激活δ珠蛋白。

IF 4 3区 医学 Q2 BIOTECHNOLOGY & APPLIED MICROBIOLOGY Human gene therapy Pub Date : 2025-04-01 Epub Date: 2025-03-21 DOI:10.1089/hum.2024.186
Lini Chen, Diandian Liu, Weicong Hong, Luhong Xu, Lin Cheng, Ying Luo, Hui Xu, Junbin Liang, Jianpei Fang, Xinyu Li
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引用次数: 0

摘要

β-地中海贫血症和镰状细胞病(SCD)是全球性的单基因血液系统疾病,重新激活的δ-球蛋白有望取代缺失或异常的β-球蛋白。随着基因编辑技术的发展,激活γ-球蛋白治疗β-地中海贫血和SCD已取得了巨大成功。然而,δ-球蛋白作为另一个重要的潜在治疗靶点,相关研究却很少。基因编辑技术将NF-Y、KLF1、GATA1和TAL1等顺式作用元件引入HBD的调控区,成功激活了δ-球蛋白的表达。研究证实,δ-球蛋白的激活效应与引入的顺式作用元件的位置密切相关。在本研究中,突变在HBD基因转录起始位点上游-85∼93 bp处创建了KLF1的全新结合位点,并在-59∼-78 bp处创建了TAL1和GATA1共结合基序的位点,这些位点可有效激活δ-球蛋白。
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Creating New Cis-Regulatory Elements of HBD to Reactivate Delta-Globin.

β-thalassemia and sickle cell disease (SCD) are global monogenic blood system disorders, and reactivated δ-globin is expected to replace missing or abnormal β-globin. With the development of gene editing technology, activating γ-globin for treating β-thalassemia and SCD has been highly successful. However, δ-globin, as another important potential therapeutic target, has few related studies. Gene editing technology introduced cis-acting elements, including NF-Y, KLF1, GATA1, and TAL1, into the regulatory region of HBD, successfully activating the expression of δ-globin. It was confirmed that the activation effect of δ-globin was closely related to the location of the introduced cis-acting elements. In this study, the mutation creates a de novo binding site for KLF1 at -85∼93 bp upstream of the transcription start site of the HBD gene, as well as the site for TAL1 and GATA1 cobinding motifs at -59 to ∼-78 bp, which could effectively activate δ-globin.

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来源期刊
Human gene therapy
Human gene therapy 医学-生物工程与应用微生物
CiteScore
6.50
自引率
4.80%
发文量
131
审稿时长
4-8 weeks
期刊介绍: Human Gene Therapy is the premier, multidisciplinary journal covering all aspects of gene therapy. The Journal publishes in-depth coverage of DNA, RNA, and cell therapies by delivering the latest breakthroughs in research and technologies. Human Gene Therapy provides a central forum for scientific and clinical information, including ethical, legal, regulatory, social, and commercial issues, which enables the advancement and progress of therapeutic procedures leading to improved patient outcomes, and ultimately, to curing diseases.
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