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First published online April 13, 2007
doi: 10.1242/10.1242/dev.02830

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Abnormal development of urogenital organs in Dlgh1-deficient mice

Akiko Iizuka-Kogo^1,*, Takefumi Ishidao^2,*,, Tetsu Akiyama^2, and Takao Senda^1,

¹ Department of Anatomy I, Fujita Health University School of Medicine, Toyoake, Aichi 470-1192, Japan.
² Laboratory of Molecular and Genetic Information, Institute for Molecular and Cellular Biosciences, The University of Tokyo, Bunkyo-ku, Tokyo 113-0032, Japan.

Author for correspondence (generation of Dlgh1 mutant mice) (e-mail: akiyama{at}iam.u-tokyo.ac.jp)

Author for correspondence (other issues) (e-mail: tsenda{at}fujita-hu.ac.jp)

Accepted 8 February 2007

Dlgh1 (discs large homolog 1) is a mammalian homolog of the Drosophila tumor suppressor Discs large 1, and is a member of the membrane-associated guanylate kinase (MAGUK) scaffolding proteins that contain three PSD-95/Dlg/ZO-1 (PDZ) domains. Discs large 1 is involved in epithelial polarization and cell-cell adhesion complex formation during Drosophila development. However, the functions of Dlgh1 during mammalian development remain to be elucidated. We generated Dlgh1-knockout mice and found that homozygous Dlgh1-knockout mice developed various abnormalities in their renal and urogenital organs. The kidneys and ureters were hypoplastic and the lower ends of the ureters were ectopic. In addition, the vagina and seminal vesicle, which are derived from the lower part of the Müllerian and Wolffian duct, respectively, were absent. Unexpectedly, loss of Dlgh1 function in the developing ureters did not disrupt cell-cell junctional complexes, but did impair cellular proliferation in the epithelium. These results suggest a novel role for Dlgh1 in regulating epithelial duct formation and morphogenesis during mammalian development. Although congenital absence of the vagina associated with other variable Müllerian duct abnormalities has been reported in humans, its mechanism has not yet been clarified. Our findings might contribute to a better understanding of such abnormalities.

Key words: Dlgh1 (Dlg1), Gene targeting, Kidney, Ureter, Vagina, Müllerian duct, Wolffian duct

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