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通过腺相关病毒介导的基因靶向和体细胞核移植生产CFTR基因缺失和CFTR-DeltaF508杂合猪。

Production of CFTR-null and CFTR-DeltaF508 heterozygous pigs by adeno-associated virus-mediated gene targeting and somatic cell nuclear transfer.

作者信息

Rogers Christopher S, Hao Yanhong, Rokhlina Tatiana, Samuel Melissa, Stoltz David A, Li Yuhong, Petroff Elena, Vermeer Daniel W, Kabel Amanda C, Yan Ziying, Spate Lee, Wax David, Murphy Clifton N, Rieke August, Whitworth Kristin, Linville Michael L, Korte Scott W, Engelhardt John F, Welsh Michael J, Prather Randall S

机构信息

Department of Internal Medicine, University of Iowa Carver College of Medicine, Iowa City, Iowa, USA.

出版信息

J Clin Invest. 2008 Apr;118(4):1571-7. doi: 10.1172/JCI34773.

Abstract

Progress toward understanding the pathogenesis of cystic fibrosis (CF) and developing effective therapies has been hampered by lack of a relevant animal model. CF mice fail to develop the lung and pancreatic disease that cause most of the morbidity and mortality in patients with CF. Pigs may be better animals than mice in which to model human genetic diseases because their anatomy, biochemistry, physiology, size, and genetics are more similar to those of humans. However, to date, gene-targeted mammalian models of human genetic disease have not been reported for any species other than mice. Here we describe the first steps toward the generation of a pig model of CF. We used recombinant adeno-associated virus (rAAV) vectors to deliver genetic constructs targeting the CF transmembrane conductance receptor (CFTR) gene to pig fetal fibroblasts. We generated cells with the CFTR gene either disrupted or containing the most common CF-associated mutation (DeltaF508). These cells were used as nuclear donors for somatic cell nuclear transfer to porcine oocytes. We thereby generated heterozygote male piglets with each mutation. These pigs should be of value in producing new models of CF. In addition, because gene-modified mice often fail to replicate human diseases, this approach could be used to generate models of other human genetic diseases in species other than mice.

摘要

由于缺乏相关动物模型,在理解囊性纤维化(CF)发病机制和开发有效治疗方法方面的进展受到了阻碍。CF小鼠不会出现导致CF患者大部分发病和死亡的肺部和胰腺疾病。猪在用于模拟人类遗传疾病方面可能比小鼠更合适,因为它们的解剖结构、生物化学、生理学、体型和遗传学与人类更为相似。然而,迄今为止,除了小鼠之外,尚未报道针对任何物种的人类遗传疾病的基因靶向哺乳动物模型。在此,我们描述了创建CF猪模型的初步步骤。我们使用重组腺相关病毒(rAAV)载体将靶向CF跨膜传导调节因子(CFTR)基因的基因构建体导入猪胎儿成纤维细胞。我们生成了CFTR基因被破坏或含有最常见CF相关突变(ΔF508)的细胞。这些细胞被用作核供体,用于将体细胞核移植到猪卵母细胞中。由此,我们生成了带有每种突变的杂合子雄性仔猪。这些猪对于创建新的CF模型具有价值。此外,由于基因修饰小鼠常常无法复制人类疾病,这种方法可用于在小鼠以外的物种中生成其他人类遗传疾病的模型。

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