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Vol. 12, No. 21, pp. 3452-3463, November 1, 1998
1 Howard Hughes Medical Institute,
2 Department of
Molecular Genetics and Cell Biology,
3 Committee on
Developmental Biology, The University of Chicago,
Chicago, Illinois 60637 USA
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.
[Key Words: ovo; skin; hair; spermatogenesis; knockout mice]
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