THE OVO GENE REQUIRED FOR CUTICLE FORMATION AND OOGENESIS IN FLIES ISINVOLVED IN HAIR FORMATION AND SPERMATOGENESIS IN MICE

The Drosophila svb/ovo gene gives rise to differentially expressed tra nscripts encoding a zinc finger protein. svb/ovo has two distinct gene tic functions: shavenbaby (svb) is required for proper formation of ex tracellular projections that are produced by certain epidermal cells i n late-stage differentiation; ovo is required for survival and differe ntiation of female germ cells. We cloned a mouse gene, movo1 encoding a nuclear transcription factor that is highly similar to its fly count erpart in its zinc-finger sequences. In mice, the gene is expressed in skin, where it localizes to the differentiating cells of epidermis an d hair follicles, and in testes, where it is present in spermatocytes and spermatids. Using gene targeting, we show that movo1 is required f or proper development of both hair and sperm. movo1(-/-) mice are smal l, 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 paralle ls between mice and flies in epidermal appendage formation and in germ -cell maturation, furthermore, they uncover a phenotype similar to tha t of Bardet-Biedl syndrome, a human disorder that maps to the same loc us as human ovo1.