Genetic Disorders – Development

Kidney International (2005) 68, 23–34; doi:10.1111/j.1523-1755.2005.00378.x

Fish mesonephric model of polycystic kidney disease in medaka (Oryzias latipes) pc mutant

EMIKO MOCHIZUKI, KATSUHIRO FUKUTA, TATSUYA TADA, TOMOHIRO HARADA, NAOKI WATANABE, SEIICHI MATSUO, HISASHI HASHIMOTO, KENJIRO OZATO and YUKO WAKAMATSU

Laboratory of Freshwater Fish Stocks, Bioscience and Biotechnology Center, Nagoya University, Nagoya, Japan; Group of Animal Organ Functions, Division of Biological Sciences, Graduate School of Science, Nagoya University, Nagoya, Japan; Laboratory of Animal Morphology and Function, Division of Biofunctions Development, Graduate School of Bioagricultural Sciences, Nagoya University Furo-cho, Chikusa-ku, Nagoya, Japan; and Department of Internal Medicine, Graduate School of Medicine, Nagoya University, Tsurumai-cho, Showa-ku, Nagoya, Japan

Correspondence: Yuko Wakamatsu, Ph.D., Laboratory of Freshwater Fish Stocks, Bioscience and Biotechnology Center, Nagoya University, Furo-cho, Chikusa-ku, Nagoya 4648601, Japan. E-mail wakamatu@bio.nagoya-u.ac.jp

Received 25 June 2004; Revised 28 January 2005; Accepted 11 February 2005.

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Abstract

Fish mesonephric model of polycystic kidney disease in medaka (Oryzias latipes) pc mutant.

Background

 

Polycystic kidney disease (PKD) is a common hereditary disease. A number of murine and zebrafish mutants have been generated and used for the study of PKD as metanephric and pronephric models, respectively. Here, we report a medaka (Oryzias latipes) mutant that develops numerous cysts in the kidney in adulthood fish in an autosomal-recessive manner as a mesonephric model of PKD.

Methods

 

The phenotypes of the medaka pc mutant were described in terms of morphologic, histologic, and ultrastructural features. The pc see-through stock was produced by crossing a pc mutant and a fish from the see-through stock and used for observing the kidney through the transparent body wall of a live fish.

Results

 

The mutant developed bilateral massive enlargement of the kidney in adulthood. They sexually matured normally within 2 months of age and died within 6 months of age. The affected kidney was occupied by numerous, fluid-filled cysts, which were lined by attenuated squamous epithelial cells. Developmentally, cystic formation began in the pronephros in 10-day-old fry and in the mesonephros in 20-day-old fry at the microscopic level. The pc see-through stock was useful in observing disease progression in live fish.

Conclusion

 

The kidney disorder that develops in the medaka pc mutant is a mesonephric counterpart of PKD, particularly an autosomal-dominant PKD, based on its morphologic, histologic, and ultrastructural features, and slow progression.

Keywords:

PKD, mesonephros, natural mutant, medaka

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