Tomonoh Yuko, Deshimaru Masanobu, Araki Kimi, Miyazaki Yasuhiro, Arasaki Tomoko, Tanaka Yasuyoshi, Kitamura Haruna, Mori Fumiaki, Wakabayashi Koichi, Yamashita Sayaka, Saito Ryo, Itoh Masayuki, Uchida Taku, Yamada Junko, Migita Keisuke, Ueno Shinya, Kitaura Hiroki, Kakita Akiyoshi, Lossin Christoph, Takano Yukio, Hirose Shinichi
Department of Pediatrics, School of Medicine, Fukuoka University, Fukuoka, Japan.
Department of Chemistry, Faculty of Science, Fukuoka University, Fukuoka, Japan.
PLoS One. 2014 Feb 19;9(2):e88549. doi: 10.1371/journal.pone.0088549. eCollection 2014.
Knock-in mouse models have contributed tremendously to our understanding of human disorders. However, generation of knock-in animals requires a significant investment of time and effort. We addressed this problem by developing a novel knock-in system that circumvents several traditional challenges by establishing stem cells with acceptor elements enveloping a particular genomic target. Once established, these acceptor embryonic stem (ES) cells are efficient at directionally incorporating mutated target DNA using modified Cre/lox technology. This is advantageous, because knock-ins are not restricted to one a priori selected variation. Rather, it is possible to generate several mutant animal lines harboring desired alterations in the targeted area. Acceptor ES cell generation is the rate-limiting step, lasting approximately 2 months. Subsequent manipulations toward animal production require an additional 8 weeks, but this delimits the full period from conception of the genetic alteration to its animal incorporation. We call this system a "kick-in" to emphasize its unique characteristics of speed and convenience. To demonstrate the functionality of the kick-in methodology, we generated two mouse lines with separate mutant versions of the voltage-dependent potassium channel Kv7.2 (Kcnq2): p.Tyr284Cys (Y284C) and p.Ala306Thr (A306T); both variations have been associated with benign familial neonatal epilepsy. Adult mice homozygous for Y284C, heretofore unexamined in animals, presented with spontaneous seizures, whereas A306T homozygotes died early. Heterozygous mice of both lines showed increased sensitivity to pentylenetetrazole, possibly due to a reduction in M-current in CA1 hippocampal pyramidal neurons. Our observations for the A306T animals match those obtained with traditional knock-in technology, demonstrating that the kick-in system can readily generate mice bearing various mutations, making it a suitable feeder technology toward streamlined phenotyping.
敲入小鼠模型对我们理解人类疾病做出了巨大贡献。然而,敲入动物的产生需要投入大量的时间和精力。我们通过开发一种新型敲入系统解决了这个问题,该系统通过建立带有包围特定基因组靶点的受体元件的干细胞,规避了几个传统挑战。一旦建立,这些受体胚胎干细胞(ES细胞)利用改良的Cre/lox技术高效地定向整合突变的靶DNA。这是有利的,因为敲入不限于一种预先选择的变异。相反,可以产生几个在靶向区域具有所需改变的突变动物品系。受体ES细胞的产生是限速步骤,持续约2个月。随后向动物生产的操作还需要8周,但这界定了从基因改变的构思到其在动物体内整合的整个时期。我们将这个系统称为“踢入”,以强调其速度和便利性的独特特征。为了证明“踢入”方法的功能,我们生成了两个带有电压依赖性钾通道Kv7.2(Kcnq2)不同突变版本的小鼠品系:p.Tyr284Cys(Y284C)和p.Ala306Thr(A306T);这两种变异都与良性家族性新生儿癫痫有关。此前未在动物中研究过的Y284C纯合成年小鼠出现自发性癫痫发作,而A306T纯合子早死。两个品系的杂合小鼠对戊四氮的敏感性增加,这可能是由于CA1海马锥体神经元中M电流减少所致。我们对A306T动物的观察结果与传统敲入技术获得的结果相符,表明“踢入”系统可以轻松地产生携带各种突变的小鼠,使其成为一种适合用于简化表型分析的饲养技术。