Zebrafish expression reporters and mutants reveal that the IgSF cell adhesion molecule Dscamb is required for feeding and survival.
Zebrafish expression reporters and mutants reveal that the IgSF cell adhesion molecule Dscamb is required for feeding and survival.
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DOI：
10.1080/01677063.2018.1493479
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发表时间：
2018-12
影响因子：
1.9
通讯作者：
Sagasti A
中科院分区：
文献类型：
--
作者：
Julien DP;Chan AW;Barrios J;Mathiaparanam J;Douglass A;Wolman MA;Sagasti A
唐氏综合征细胞粘附分子（DSCAMs）在神经系统中广泛表达，并在程序性细胞死亡、神经元迁移、轴突导向、神经突分支和间隔以及突触靶向中发挥保守作用。然而，DSCAM似乎在不同的脊椎动物中具有不同的功能，并且对其在视网膜外的功能知之甚少。我们利用斑马鱼幼鱼的遗传易处理性和光学可及性来研究DSCAM家族成员dscamb的表达和功能。使用靶向基因组编辑来创建转基因报告基因和功能丧失突变等位基因，我们发现dscamb在整个大脑，脊髓和周围神经系统中广泛表达，但不是大脑整体结构组织所必需的。尽管没有明显的解剖缺陷，纯合子dscamb突变体缺乏摄取食物的能力，很少存活到成年。因此，我们发现了dscamb在摄食行为中的新功能。在这些研究中产生的突变体和转基因株系将为鉴定这些行为的分子和细胞基础提供有价值的工具。
Down Syndrome Cell Adhesion Molecules (DSCAMs) are broadly expressed in nervous systems and play conserved roles in programmed cell death, neuronal migration, axon guidance, neurite branching and spacing, and synaptic targeting. However, DSCAMs appear to have distinct functions in different vertebrate animals, and little is known about their functions outside the retina. We leveraged the genetic tractability and optical accessibility of larval zebrafish to investigate the expression and function of a DSCAM family member, dscamb. Using targeted genome editing to create transgenic reporters and loss-of-function mutant alleles, we discovered that dscamb is expressed broadly throughout the brain, spinal cord, and peripheral nervous system, but is not required for overall structural organization of the brain. Despite the absence of obvious anatomical defects, homozygous dscamb mutants were deficient in their ability to ingest food and rarely survived to adulthood. Thus, we have discovered a novel function for dscamb in feeding behavior. The mutant and transgenic lines generated in these studies will provide valuable tools for identifying the molecular and cellular bases of these behaviors.
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影响因子：
--
作者：
Diogo R;Hinits Y;Hughes SM
通讯作者：
Hughes SM
影响因子：
16.2
作者：
Fuerst, Peter G.;Bruce, Freyja;Tian, Miao;Wei, Wei;Elstrott, Justin;Feller, Maria B.;Erskine, Lynda;Singer, Joshua H.;Burgess, Robert W.
通讯作者：
Burgess, Robert W.
影响因子：
4.5
作者：
Das A;Crump JG
通讯作者：
Crump JG
DOI：
10.1006/bbrc.2001.5214
发表时间：
2001-07-20
影响因子：
3.1
作者：
Agarwala, KL;Ganesh, S;Yamakawa, K
通讯作者：
Yamakawa, K
DOI：
10.1016/s0006-291x(02)00307-8
发表时间：
2002-05-10
影响因子：
3.1
作者：
Barlow, GM;Micales, B;Korenberg, JR
通讯作者：
Korenberg, JR