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利用 CRISPR-Cas9 外显子缺失策略生产杜氏肌营养不良症细胞模型。

Production of Duchenne muscular dystrophy cellular model using CRISPR-Cas9 exon deletion strategy.

机构信息

Department of Medical Genetics and Molecular Medicine, School of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.

Student Research Committee, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran.

出版信息

Mol Cell Biochem. 2024 May;479(5):1027-1040. doi: 10.1007/s11010-023-04759-3. Epub 2023 Jun 8.

DOI:10.1007/s11010-023-04759-3
PMID:37289342
Abstract

Duchenne Muscular Dystrophy (DMD) is a progressive muscle wasting disorder caused by loss-of-function mutations in the dystrophin gene. Although the search for a definitive cure has failed to date, extensive efforts have been made to introduce effective therapeutic strategies. Gene editing technology is a great revolution in biology, having an immediate application in the generation of research models. DMD muscle cell lines are reliable sources to evaluate and optimize therapeutic strategies, in-depth study of DMD pathology, and screening the effective drugs. However, only a few immortalized muscle cell lines with DMD mutations are available. In addition, obtaining muscle cells from patients also requires an invasive muscle biopsy. Mostly DMD variants are rare, making it challenging to identify a patient with a particular mutation for a muscle biopsy. To overcome these challenges and generate myoblast cultures, we optimized a CRISPR/Cas9 gene editing approach to model the most common DMD mutations that include approximately 28.2% of patients. GAP-PCR and sequencing results show the ability of the CRISPR-Cas9 system to efficient deletion of mentioned exons. We showed producing truncated transcript due to the targeted deletion by RT-PCR and sequencing. Finally, mutation-induced disruption of dystrophin protein expression was confirmed by western blotting. All together, we successfully created four immortalized DMD muscle cell lines and showed the efficacy of the CRISPR-Cas9 system for the generation of immortalized DMD cell models with the targeted deletions.

摘要

杜氏肌营养不良症(DMD)是一种进行性肌肉消耗疾病,由肌营养不良蛋白基因突变引起功能丧失。尽管迄今为止尚未找到明确的治愈方法,但已经做出了广泛的努力来引入有效的治疗策略。基因编辑技术是生物学的一场重大革命,它在研究模型的产生方面具有直接的应用。DMD 肌肉细胞系是评估和优化治疗策略、深入研究 DMD 病理学以及筛选有效药物的可靠来源。然而,仅有少数带有 DMD 突变的永生化肌肉细胞系可用。此外,从患者中获取肌肉细胞还需要进行有创的肌肉活检。大多数 DMD 变体都很罕见,因此很难为肌肉活检确定特定突变的患者。为了克服这些挑战并生成成肌细胞培养物,我们优化了 CRISPR/Cas9 基因编辑方法,以模拟包括约 28.2%的患者在内的最常见的 DMD 突变。GAP-PCR 和测序结果表明,CRISPR-Cas9 系统能够有效地删除这些外显子。我们通过 RT-PCR 和测序显示了由于靶向缺失而产生截断转录本。最后,通过 Western blot 证实了突变诱导的肌营养不良蛋白表达中断。总之,我们成功创建了四个永生化 DMD 肌肉细胞系,并展示了 CRISPR-Cas9 系统在生成具有靶向缺失的永生化 DMD 细胞模型方面的有效性。

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New advancements in CRISPR based gene therapy of Duchenne muscular dystrophy.基于 CRISPR 的杜氏肌营养不良症基因治疗的新进展。
Gene. 2023 May 30;867:147358. doi: 10.1016/j.gene.2023.147358. Epub 2023 Mar 11.
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Efficient correction of Duchenne muscular dystrophy mutations by SpCas9 and dual gRNAs.利用SpCas9和双gRNA高效校正杜氏肌营养不良症突变
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Detailed genetic and functional analysis of the hDMDdel52/mdx mouse model.
详细的 hDMDdel52/mdx 小鼠模型的遗传和功能分析。
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Correction of Three Prominent Mutations in Mouse and Human Models of Duchenne Muscular Dystrophy by Single-Cut Genome Editing.通过单切基因组编辑纠正杜氏肌营养不良症小鼠和人类模型中的三个突出突变。
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Cells. 2020 Apr 2;9(4):869. doi: 10.3390/cells9040869.
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