SUBTLE CEREBELLAR PHENOTYPE IN MICE HOMOZYGOUS FOR A TARGETED DELETION OF THE EN-2 HOMEOBOX

SUBTLE CEREBELLAR PHENOTYPE IN MICE HOMOZYGOUS FOR A TARGETED DELETION OF THE EN-2 HOMEOBOX
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DOI:
10.1126/science.1672471
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发表时间:
1991-03-08
期刊:
影响因子:
56.9
通讯作者:
ROSSANT, J
ROSSANT, J
中科院分区:
综合性期刊1区
文献类型:
--
作者:
JOYNER, AL;HERRUP, K;ROSSANT, J

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小鼠的两个基因 En-1 和 En-2 是果蝇分割基因的同源物,在胚胎发生过程中在神经管中表现出重叠的空间限制表达模式,表明在区域规范中发挥作用。删除同源盒的靶向突变的纯合小鼠是可行的,并且在胚胎发育中没有表现出明显的缺陷。这可能是由于胚胎发生过程中 En-2 和相关 En-1 基因产物的功能冗余所致。与这一假设一致的是,突变小鼠在成年小脑中表现出异常的叶状结构,其中正常表达 En-2,而不是 En-1。
The two mouse genes, En-1 and En-2, that are homologs of the Drosophila segmentation gene engrailed, show overlapping spatially restricted patterns of expression in the neural tube during embryogenesis, suggestive of a role in regional specification. Mice homozygous for a targeted mutation that deletes the homeobox were viable and showed no obvious defects in embryonic development. This may be due to functional redundancy of En-2 and the related En-1 gene product during embryogenesis. Consistent with this hypothesis, the mutant mice showed abnormal foliation in the adult cerebellum, where En-2, and not En-1, is normally expressed.