brother of cdo (umleitung) is cell-autonomously required for Hedgehog-mediated ventral CNS patterning in the zebrafish

brother of cdo (umleitung) is cell-autonomously required for Hedgehog-mediated ventral CNS patterning in the zebrafish
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DOI:
10.1242/dev.057950
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发表时间:
2011-01-01
期刊:
影响因子:
4.6
通讯作者:
Karlstrom, Rolf O.
Karlstrom, Rolf O.
中科院分区:
生物学2区
文献类型:
--
作者:
Bergeron, Sadie A.;Tyurina, Oksana V.;Karlstrom, Rolf O.

文献摘要

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CDO的跨膜蛋白Brother of CDO(Boc)参与Shh介导的连合轴突引导,可以正向和负向调节Hedgehog(HH)靶基因的转录,但对Boc在脊椎动物胚胎发育中的体内需求知之甚少。斑马鱼umleitung(umleitung,UM(Ty54))突变体通过视网膜顶盖轴突投射缺陷进行鉴定。在这里,我们证明了UMLLocus编码Boc,并且Boc功能是细胞自主地需要的,HH介导的神经模式。我们的表型分析表明,Boc是脊髓、下丘脑、垂体、体节和上颌中HH信号的正向调节因子,但Boc可能对下颌骨中的HH信号进行负向调节。这项研究揭示了Boc在接受高水平HH的腹侧CNS细胞中的作用,并揭示了Boc在脊椎动物胚胎发生中先前未知的角色。
The transmembrane protein Brother of Cdo (Boc) has been implicated in Shh-mediated commissural axon guidance, and can both positively and negatively regulate Hedgehog (Hh) target gene transcription, however, little is known about in vivo requirements for Boc during vertebrate embryogenesis. The zebrafish umleitung (uml(ty54)) mutant was identified by defects in retinotectal axon projections. Here, we show that the uml locus encodes Boc and that Boc function is cell-autonomously required for Hh-mediated neural patterning. Our phenotypic analysis suggests that Boc is required as a positive regulator of Hh signaling in the spinal cord, hypothalamus, pituitary, somites and upper jaw, but that Boc might negatively regulate Hh signals in the lower jaw. This study reveals a role for Boc in ventral CNS cells that receive high levels of Hh and uncovers previously unknown roles for Boc in vertebrate embryogenesis.