A genetic screen for zebrafish mutants with hepatic steatosis identifies a locus required for larval growth.

A genetic screen for zebrafish mutants with hepatic steatosis identifies a locus required for larval growth.
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DOI:
10.1111/joa.12570
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发表时间:
2017-03
期刊:
影响因子:
2.4
通讯作者:
Schlegel A
Schlegel A
中科院分区:
医学3区
文献类型:
--
作者:
Hugo SE;Schlegel A

文献摘要

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在筛选肝脏脂质过度积累(肝脂肪变性)的斑马鱼幼虫突变体时,我们发现了收获月(hmn)。 hmn 突变肝细胞中可见细胞质脂滴,周围有多囊泡结构和嵴肿胀的线粒体。 hmn 突变幼虫全身三酰甘油增加。当我们尝试将在筛选的发育阶段形态正常的突变体饲养到成年时,我们观察到大多数 hmn 突变体在饲养时无法存活到幼年期。生长停滞发生在幼虫后期,没有明显的器官缺陷。母本合子突变体没有额外的缺陷,表明该突变影响后期发育过程。胚胎发生和变态之间的发育窗口仍未得到充分研究,hmn 突变体可能有助于探索这一过渡时期发生的分子和解剖过程。
In a screen for zebrafish larval mutants with excessive liver lipid accumulation (hepatic steatosis), we identified harvest moon (hmn). Cytoplasmic lipid droplets, surrounded by multivesicular structures and mitochondria whose cristae appeared swollen, are seen in hmn mutant hepatocytes. Whole body triacylglycerol is increased in hmn mutant larvae. When we attempted to raise mutants, which were morphologically normal at the developmental stage that the screen was conducted, to adulthood, we observed that most hmn mutants do not survive to the juvenile period when raised. An arrest in growth occurs in the late larval period without obvious organ defects. Maternal zygotic mutants have no additional defects, suggesting that the mutation affects a late developmental process. The developmental window between embryogenesis and the metamorphosis remains under-studied, and hmn mutants might be useful for exploring the molecular and anatomic processes occurring during this transition period.