Behavioral analysis of male and female Fmr1 knockout mice on C57BL/6 background.
Behavioral analysis of male and female Fmr1 knockout mice on C57BL/6 background.
复制标题
在C57BL/6背景上对男性和女性FMR1敲除小鼠的行为分析。
DOI：
10.1016/j.bbr.2014.05.046
复制
发表时间：
2014-09-01
影响因子：
2.7
通讯作者：
Wang, Hongbing
中科院分区：
文献类型：
--
作者：
Ding, Qi;Sethna, Ferzin;Wang, Hongbing
脆性X综合征（FXS）是由FMR 1基因突变引起的单基因疾病。Fmr 1基因敲除（KO）小鼠显示出FXS相关表型的许多方面，并已被用作FXS的主要临床前模型。虽然FXS发生在男性和女性患者中，但大多数关于小鼠模型的研究使用雄性动物。很少有研究测试性别是否会影响小鼠模型的表面有效性。在这里，我们研究了C57 BL/6背景下雄性半合子和雌性纯合子Fmr 1 KO小鼠的多种行为表型。对于每种行为范式，我们检查了来自不同窝的多个队列。我们发现，男性和女性的Fmr 1基因敲除小鼠表现出显着的听源性癫痫发作，多动在开放领域的测试，赤字在被动回避和上下文的恐惧记忆，并显着增强PPI在低刺激强度。雄性和雌性Fmr 1 KO小鼠在亮-暗试验中也显示出更多的亮室和暗室之间的过渡运动。缺乏性别效应表明，Fmr 1 KO小鼠是测试潜在FXS疗法疗效的合理工具。
Fragile X syndrome (FXS) is a monogenic disease caused by mutations in the FMR1 gene. The Fmr1 knockout (KO) mice show many aspects of FXS-related phenotypes, and have been used as a major pre-clinical model for FXS. Although FXS occurs in both male and female patients, most studies on the mouse model use male animals. Few studies test whether gender affects the face validity of the mouse model. Here, we examined multiple behavioral phenotypes with male hemizygous and female homozygous Fmr1 KO mice on C57BL/6 background. For each behavioral paradigm, we examined multiple cohorts from different litters. We found that both male and female Fmr1 KO mice displayed significant audiogenic seizures, hyperactivity in the open field test, deficits in passive avoidance and contextual fear memory, and significant enhancement of PPI at low stimulus intensity. Male and female Fmr1 KO mice also showed more transitional movement between the lit and dark chambers in the light-dark tests. The lack of gender effects suggests that the Fmr1 KO mouse is a reasonable tool to test the efficacy of potential FXS therapies.
登录
查看更多内容
影响因子：
16.2
作者：
Edbauer, Dieter;Neilson, Joel R.;Foster, Kelly A.;Wang, Chi-Fong;Seeburg, Daniel P.;Batterton, Matthew N.;Tada, Tomoko;Dolan, Bridget M.;Sharp, Phillip A.;Sheng, Morgan
通讯作者：
Sheng, Morgan
DOI：
10.1111/j.1601-183x.2012.00781.x
发表时间：
2012-07
期刊：
Genes, brain, and behavior
影响因子：
--
作者：
Goebel-Goody SM;Wilson-Wallis ED;Royston S;Tagliatela SM;Naegele JR;Lombroso PJ
通讯作者：
Lombroso PJ
影响因子：
3.3
作者：
Qin, M.;Xia, Z.;Huang, T.;Smith, C. B.
通讯作者：
Smith, C. B.
DOI：
10.1073/pnas.242377399
发表时间：
2002-11-26
影响因子：
11.1
作者：
Qin, M;Kang, J;Smith, CB
通讯作者：
Smith, CB
DOI：
10.1523/jneurosci.0402-10.2010
发表时间：
2010-08-11
期刊：
The Journal of neuroscience : the official journal of the Society for Neuroscience
影响因子：
--
作者：
Gross C;Nakamoto M;Yao X;Chan CB;Yim SY;Ye K;Warren ST;Bassell GJ
通讯作者：
Bassell GJ