The ovo gene required for cuticle formation and oogenesis in flies is involved in hair formation and spermatogenesis in mice

Abstract

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.

Ovol2, a Mammalian Homolog of Drosophila ovo: Gene Structure, Chromosomal Mapping, and Aberrant Expression in Blind-Sterile Mice

“…So far, three ovo family members have been functionally characterized. Drosophila melanogaster ovo and mouse Ovol1 (also known as movo1) are required for germ cell and epidermal appendage differentiation [2][3][4][5][6], whereas mutations in Caenorhabditis elegans ovo do not produce apparent fertility or epidermal defects, but instead cause defects in hindgut (the counterpart of mammalian urogenital system, where defects are found in Ovol1 mutant mice) [7].…”

“…Two of the transcripts encode the same nuclear protein that binds DNA sequences similar to the Drosophila OVO recognition site [1], whereas the complete sequence of the third and largest transcript remains to be determined. All three Ovol1 transcripts are detected in mouse testis, specifically in premeiotic and meiotic germ cells, and in skin, specifically in differentiating cells of the epidermis and hair follicles [6]. Ablation of Ovol1 in mice leads to male germ cell degeneration and structural abnormalities in the hair shafts, but no obvious defect in the epidermis [6].…”

“…All three Ovol1 transcripts are detected in mouse testis, specifically in premeiotic and meiotic germ cells, and in skin, specifically in differentiating cells of the epidermis and hair follicles [6]. Ablation of Ovol1 in mice leads to male germ cell degeneration and structural abnormalities in the hair shafts, but no obvious defect in the epidermis [6]. Although these results support the model that ovo function in germ cell and epidermal appendage differentiation is conserved from flies to mice, they likely do not uncover the full spectrum of ovo functions in mammals, as a single fly gene usually evolves into multiple mammalian homologs, and some of the defects of Drosophila ovo mutants (such as those observed in the female germ line) have not been observed in mice deficient for Ovol1.…”

“…A second mouse ovo gene, Ovol2 (also known as movo2) [6], exists, and cloning of two Ovol2 cDNAs from a mouse testis library was reported [14]. Here we provide a corrected version of the mouse Ovol2 cDNA sequences; report the isolation, characterization, and chromosomal localization of the mouse Ovol2 gene; compare mouse and human OVOL2 structure and tissue expression; and present evidence that mouse Ovol2 is expressed in male germ cells and that this expression is developmentally regulated.…”

09/15: Comparative Genomics of a Conserved Chromosomal Region Associated with a Complex Human Phenotype

“…The physical mapping of all genes within this interval will now allow us to understand how closely spaced genes are differentially regulated, and how enhancer activities are insulated from each other. Interestingly, a phenotype resembling aspects of Bardet-Biedl Syndrome was found in mice that carry a targeted disruption of the Ovo1 gene, which encodes a Zinc-finger transcription factor (Dai et al, 1998). Assuming that genes on different Bardet-Biedl loci act in the same developmental and cellular pathways, it is conceivable that genes in the 09/15 synteny region could be regulated by this transcription factor.…”