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首页> 外文期刊>Molecular and Cellular Biology >Dach1 Mutant Mice Bear No Gross Abnormalities in Eye, Limb, and Brain Development and Exhibit Postnatal Lethality
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Dach1 Mutant Mice Bear No Gross Abnormalities in Eye, Limb, and Brain Development and Exhibit Postnatal Lethality

机译：Dach1突变小鼠的眼睛，肢体和大脑发育没有明显异常，并且显示出产后致死率

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摘要

Drosophila dachshund is necessary and sufficient for compound eye development and is required for normal leg and brain development. A mouse homologue of dachshund, Dach1, is expressed in the developing retina and limbs, suggesting functional conservation of this gene. We have generated a loss-of-function mutation in Dach1 that results in the abrogation of the wild-type RNA and protein expression pattern in embryos. Homozygous mutants survive to birth but exhibit postnatal lethality associated with a failure to suckle, cyanosis, and respiratory distress. The heart, lungs, kidneys, liver, and skeleton were examined to identify factors involved in postnatal lethality, but these organs appeared to be normal. In addition, blood chemistry tests failed to reveal differences that might explain the lethal phenotype. Gross examination and histological analyses of newborn eyes, limbs, and brains revealed no detectable abnormalities. Since Dach1 mutants die shortly after birth, it remains possible thatDach1 is required for postnatal development of these structures. Alternatively, an additional Dach homologue may functionally compensate for Dach1 loss of function.
机译：果蝇腊肠对于复眼发育是必需的，并且对于正常的腿部和大脑发育是必需的。达克斯猎犬，Dach1 的小鼠同源物在发育中的视网膜和四肢中表达，表明该基因的功能保守。我们在 Dach1 中产生了功能丧失突变，导致胚胎中的野生型RNA和蛋白质表达模式被取消。纯合突变体可以存活到出生，但是表现出与哺乳，紫和呼吸窘迫失败相关的产后致死率。检查了心脏，肺，肾脏，肝脏和骨骼，以确定与出生后致死率有关的因素，但这些器官似乎正常。此外，血液化学测试未能揭示可能解释致死表型的差异。新生儿眼睛，四肢和大脑的粗略检查和组织学分析未发现可检测到的异常。由于 Dach1 突变体出生后不久就会死亡，因此 Dach1 仍可能是这些结构的出生后发育所必需的。或者，其他 Dach 同源物可以在功能上补偿 Dach1 的功能丧失。

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