Decreased home cage movement and oromotor impairments in adult Fmr1-KO mice.
Decreased home cage movement and oromotor impairments in adult Fmr1-KO mice.
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DOI：
10.1111/gbb.12374
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发表时间：
2017-06
期刊：
影响因子：
--
通讯作者：
Dunaevsky A
中科院分区：
文献类型：
--
作者：
Bonasera SJ;Chaudoin TR;Goulding EH;Mittek M;Dunaevsky A
脆性 X 综合征 (FXS) 是一种常见的遗传性疾病，会严重影响家庭和患者整个生命周期的日常生活。 FXS 对儿童和青少年行为的影响已广为人知。然而，评估 FXS 成人行为表型的研究（除了那些检查精神合并症的研究）明显较少。带有 Fmr1 基因损伤的小鼠重现了 FXS 的重要分子和神经解剖学特征，并提供了一种评估与 FXS 相关的成年行为表型的方法。我们首次描述了基线行为，包括进食、饮水、运动及其昼夜节律；在成年 Fmr1-KO 突变小鼠的广泛环境适应后连续 16 天观察到所有这些。我们发现小鼠的食物摄入、进食模式、新陈代谢或运动、进食和饮水的昼夜节律模式没有基因型变化。适应后，Fmr1-KO 小鼠在活跃期（暗周期）的日常运动明显减少。然而，与野生型相比，Fmr1-KO 小鼠在光周期中有更多的活动。此外，Fmr1-KO小鼠在昼夜节律黑暗周期中每天摄入的水量显着减少，并且这种水摄入量的减少伴随着每次舔水量的减少。在 Fmr1-KO 小鼠中观察到的水摄入和昼夜节律表型概括了先前在 FXS 中描述的已知临床方面。 Fmr1-KO 小鼠运动减少的发现是新颖的，这表明 Fmr1-KO 小鼠的基线和新奇诱发的活动之间存在分离。
Fragile X syndrome (FXS) is a common inherited disorder that significantly impacts family and patient day-to-day living across the entire lifespan. The childhood and adolescent behavioral consequences of FXS are well-appreciated. However, there are significantly fewer studies (except those examining psychiatric comorbidities) assessing behavioral phenotypes seen in adults with FXS. Mice engineered with a genetic lesion of Fmr1 recapitulate important molecular and neuroanatomical characteristics of FXS, and provide a means to evaluate adult behavioral phenotypes associated with FXS. We give the first description of baseline behaviors including feeding, drinking, movement, and their circadian rhythms; all observed over 16 consecutive days following extensive environmental habituation in adult Fmr1-KO mutant mice. We find no genotypic changes in mouse food ingestion, feeding patterns, metabolism, or circadian patterns of movement, feeding, and drinking. After habituation, Fmr1-KO mice demonstrate significantly less daily movement during their active phase (the dark cycle). However, Fmr1-KO mice have more bouts of activity during the light cycle compared to wildtypes. In addition, Fmr1-KO mice demonstrate significantly less daily water ingestion during the circadian dark cycle, and this reduction in water intake is accompanied by a decrease in the amount of water ingested per lick. The observed water ingestion and circadian phenotypes noted in Fmr1-KO mice recapitulate known clinical aspects previously described in FXS. The finding of decreased movement in Fmr1-KO mice is novel, and suggests a dissociation between baseline and novelty-evoked activity for Fmr1-KO mice.
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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：
5.7
作者：
Ellegood, Jacob;Pacey, Laura K.;Henkelman, R. Mark
通讯作者：
Henkelman, R. Mark
影响因子：
7.6
作者：
Gholizadeh, Shervin;Arsenault, Jason;Hampson, David R.
通讯作者：
Hampson, David R.
影响因子：
30.8
作者：
DEVYS, D;LUTZ, Y;MANDEL, JL
通讯作者：
MANDEL, JL
DOI：
10.1352/1944-7558-116.1.16
发表时间：
2011-01-01
影响因子：
2.1
作者：
Hartley, Sigan L.;Seltzer, Marsha Mailick;Bailey, Donald B., Jr.
通讯作者：
Bailey, Donald B., Jr.