Foxg1 deletion impairs the development of the epithalamus.

Foxg1 deletion impairs the development of the epithalamus.
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Foxg1缺失损害上丘脑的发育

DOI:
10.1186/s13041-018-0350-2
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发表时间:
2018-02-02
期刊:
影响因子:
3.6
通讯作者:
Zhao C
Zhao C
中科院分区:
医学3区
文献类型:
--
作者:
Liu B;Zhou K;Wu X;Zhao C

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上皮层位于丘脑的背侧,由缰核、松果体和第三脑室脉络丛组成,在脊椎动物的应激反应和睡眠-觉醒周期中发挥重要作用。在发育过程中,上皮层起源于前体2的最背部。然而,上皮层发育的机制在很大程度上仍不清楚。FOXG1对端脑的发育至关重要,但它在间脑发育中的作用一直未得到充分的研究。患有FOXG1相关疾病的患者表现出严重的焦虑、睡眠障碍和脉络丛囊肿,表明FOXG1可能在上皮性发育中发挥作用。在本研究中,我们鉴定了FOXG1在发育中的上皮细胞中的特异性表达。使用“自我删除”的方法,我们发现缰核显著扩张,包括数量增加的缰核亚型神经元。神经,特别是缰连合部严重受损。同时,FOXG1突变体的松果体减少,脉络丛分枝增多。FOXG1消融后,Shh和FGFs信号无明显变化,提示FOXG1不参与Shh和FGFs参与调节上皮层的发育。我们的发现为调节上皮层的发育提供了新的见解。
The epithalamus, which is dorsal to the thalamus, consists of the habenula, pineal gland and third ventricle choroid plexus and plays important roles in the stress response and sleep–wake cycle in vertebrates. During development, the epithalamus arises from the most dorsal part of prosomere 2. However, the mechanism underlying epithalamic development remains largely unknown. Foxg1 is critical for the development of the telencephalon, but its role in diencephalic development has been under-investigated. Patients suffering from FOXG1-related disorders exhibit severe anxiety, sleep disturbance and choroid plexus cysts, indicating that Foxg1 likely plays a role in epithalamic development. In this study, we identified the specific expression of Foxg1 in the developing epithalamus. Using a “self-deletion” approach, we found that the habenula significantly expanded and included an increased number of habenular subtype neurons. The innervations, particularly the habenular commissure, were severely impaired. Meanwhile, the Foxg1 mutants exhibited a reduced pineal gland and more branched choroid plexus. After ablation of Foxg1 no obvious changes in Shh and Fgf signalling were observed, suggesting that Foxg1 regulates the development of the epithalamus without the involvement of Shh and Fgfs. Our findings provide new insights into the regulation of the development of the epithalamus.
FOXG1 综合征的核心表型包括出生后小头畸形、严重智力低下、语言缺失、运动障碍和胼胝体发育不全。
DOI: 10.1136/jmg.2010.087528
发表时间: 2011-06
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期刊: DEVELOPMENT
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DOI: 10.1016/0306-4522(87)90337-x
发表时间: 1987-04-01
期刊: NEUROSCIENCE
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