Dach1 mutant mice bear no gross abnormalities in eye, limb, and brain development and exhibit postnatal lethality
Dach1 mutant mice bear no gross abnormalities in eye, limb, and brain development and exhibit postnatal lethality
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DOI：
10.1128/mcb.21.5.1484-1490.2001
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发表时间：
2001-03-01
影响因子：
5.3
通讯作者：
Mardon, G
中科院分区：
文献类型：
--
作者：
Davis, RJ;Shen, WP;Mardon, G
Drosophila dachshund是复眼发育所必需的，也是正常腿和大脑发育所必需的。腊肠犬的小鼠同源物Dach 1在发育中的视网膜和四肢中表达，表明该基因的功能保守。我们已经在Dach 1中产生了一个功能缺失突变，导致胚胎中野生型RNA和蛋白质表达模式的废除。纯合子突变体存活到出生，但表现出出生后的致命性与失败，发绀？和呼吸窘迫心脏、肺、肾、肝和骨骼被检查以确定与出生后死亡有关的因素，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、四肢和大脑的大体检查和组织学分析未发现异常。因为Dach 1变种人死了！出生后不久，Dach 1可能是这些结构出生后发育所必需的。或者，额外的Dach同源物可以在功能上补偿Dach 1功能的丧失。
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, hut 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 re mains possible that Dach1 is required for postnatal development of these structures.;Alternatively, an additional Dach homologue may functionally compensate for Dach1 loss of function.