The power of zebrafish models for understanding the co-occurrence of craniofacial and limb disorders.
The power of zebrafish models for understanding the co-occurrence of craniofacial and limb disorders.
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斑马鱼模型在理解头面部和四肢疾病并存方面的能力。
DOI：
10.1002/dvg.23407
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发表时间：
2021-03
期刊：
影响因子：
--
通讯作者：
Artinger KB
中科院分区：
文献类型：
--
作者：
Truong BT;Artinger KB
颅面和肢体缺损是普通人群中最常见的两种先天性畸形。有趣的是，这些缺陷并不相互排斥。许多颅面表型的患者，如口面裂和颅缝闭合，也存在肢体缺陷，包括多指、并指、短指或外指。控制颅面和肢体发育的基因调控网络最初似乎彼此不同，然而这些出生缺陷经常同时发生。这两个发育过程在脊椎动物中都是高度保守的，而斑马鱼由于其高繁殖力、相对容易的基因操作和发育过程的透明性而成为一种有利的模型。在这里，我们总结了使用斑马鱼模型研究具有颅面和肢体表型的人类综合征的研究。我们讨论了颅面和肢体/鳍发育的高度保守过程，并描述了最近的斑马鱼研究，这些研究探索了在这两种结构中与人类综合征表型相关的基因功能。我们试图找出两者之间的共性，以帮助解释为什么颅面和肢体异常经常一起发生。
Craniofacial and limb defects are two of the most common congenital anomalies in the general population. Interestingly, these defects are not mutually exclusive. Many patients with craniofacial phenotypes, such as orofacial clefting and craniosynostosis, also present with limb defects, including polydactyly, syndactyly, brachydactyly, or ectrodactyly. The gene regulatory networks governing craniofacial and limb development initially seem distinct from one another, and yet these birth defects frequently occur together. Both developmental processes are highly conserved among vertebrates, and zebrafish have emerged as an advantageous model due to their high fecundity, relative ease of genetic manipulation, and transparency during development. Here we summarize studies that have used zebrafish models to study human syndromes that present with both craniofacial and limb phenotypes. We discuss the highly conserved processes of craniofacial and limb/fin development and describe recent zebrafish studies that have explored the function of genes associated with human syndromes with phenotypes in both structures. We attempt to identify commonalities between the two to help explain why craniofacial and limb anomalies often occur together.
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影响因子：
2
作者：
Clouthier, David E.;Garcia, Elvin;Schilling, Thomas F.
通讯作者：
Schilling, Thomas F.
影响因子：
4.5
作者：
Das A;Crump JG
通讯作者：
Crump JG
影响因子：
4.6
作者：
Dougherty, Max;Kamel, George;Liao, Eric C.
通讯作者：
Liao, Eric C.
斑马鱼裂口的模型。
DOI：
10.1002/dvdy.24566
发表时间：
2017-11
期刊：
Developmental dynamics : an official publication of the American Association of Anatomists
影响因子：
--
作者：
Duncan KM;Mukherjee K;Cornell RA;Liao EC
通讯作者：
Liao EC
影响因子：
11.8
作者：
Bakkers, J;Hild, M;Hammerschmidt, M
通讯作者：
Hammerschmidt, M