The ovo gene required for cuticle formation and oogenesis in flies is involved in hair formation and spermatogenesis in mice
The ovo gene required for cuticle formation and oogenesis in flies is involved in hair formation and spermatogenesis in mice
复制标题
DOI：
10.1101/gad.12.21.3452
复制
发表时间：
1998-11-01
影响因子：
10.5
通讯作者：
Fuchs, E
中科院分区：
文献类型：
--
作者：
Dai, X;Schonbaum, C;Fuchs, E
果蝇svb/ovo基因产生编码锌指蛋白的差异表达转录本。Svb /ovo具有两种不同的遗传功能：剃须婴儿（Svb）是某些表皮细胞在分化后期产生的细胞外突起的正确形成所必需的；卵细胞是雌性生殖细胞存活和分化所必需的。我们克隆了一个小鼠基因，movo1编码一个核转录因子，其锌指序列与苍蝇的核转录因子高度相似。在小鼠中，该基因在皮肤中表达，它定位于表皮和毛囊的分化细胞，在睾丸中表达，它存在于精母细胞和精子细胞中。通过基因靶向，我们发现movo1是头发和精子正常发育所必需的。Movo1（-/-）小鼠体型小，毛发异常，表现为生殖能力低下。这些小鼠的肾脏也出现异常，其中也表达了movo1。我们的发现揭示了小鼠和苍蝇在表皮附属物形成和生殖细胞成熟方面的显著相似之处，此外，他们发现了一种与Bardet-Biedl综合征相似的表型，Bardet-Biedl综合征是一种人类疾病，与人类ovo1位于相同的位点。
The Drosophila svb/ovo gene gives rise to differentially expressed transcripts encoding a zinc finger protein. svb/ovo has two distinct genetic functions: shavenbaby (svb) is required for proper formation of extracellular projections that are produced by certain epidermal cells in late-stage differentiation; ovo is required for survival and differentiation of female germ cells. We cloned a mouse gene, movo1 encoding a nuclear transcription factor that is highly similar to its fly counterpart in its zinc-finger sequences. In mice, the gene is expressed in skin, where it localizes to the differentiating cells of epidermis and hair follicles, and in testes, where it is present in spermatocytes and spermatids. Using gene targeting, we show that movo1 is required for proper development of both hair and sperm. movo1(-/-) mice are small, produce aberrant hairs, and display hypogenitalism, with a reduced ability to reproduce. These mice also develop abnormalities in kidney, where movo1 is also expressed. Our findings reveal remarkable parallels between mice and flies in epidermal appendage formation and in germ-cell maturation, furthermore, they uncover a phenotype similar to that of Bardet-Biedl syndrome, a human disorder that maps to the same locus as human ovo1.