title: Fish mesonephric model of polycystic kidney disease in medaka (Oryzias latipes) pc mutant.
abstract: 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.
  
  
  CONCLUSIONS
  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.
authors.affiliation: Laboratory of Freshwater Fish Stocks, Bioscience and Biotechnology Center, Nagoya University, Nagoya, Japan.
authors.forename: Emiko
authors.initials: E
authors.lastname: Mochizuki
authors.forename: Katsuhiro
authors.initials: K
authors.lastname: Fukuta
authors.forename: Tatsuya
authors.initials: T
authors.lastname: Tada
authors.forename: Tomohiro
authors.initials: T
authors.lastname: Harada
authors.forename: Naoki
authors.initials: N
authors.lastname: Watanabe
authors.forename: Seiichi
authors.initials: S
authors.lastname: Matsuo
authors.forename: Hisashi
authors.initials: H
authors.lastname: Hashimoto
authors.forename: Kenjiro
authors.initials: K
authors.lastname: Ozato
authors.forename: Yuko
authors.initials: Y
authors.lastname: Wakamatsu
mesh_terms: D000818:Animals; D004195:Disease Models, Animal; D005260:Female; D005799:Genes, Dominant; D007684:Kidney Tubules / Q000002:abnormalities / Q000473:pathology / Q000648:ultrastructure; D008297:Male; D008650:Mesonephros / Q000473:pathology* / Q000648:ultrastructure; D008855:Microscopy, Electron, Scanning; D046529:Microscopy, Electron, Transmission; D009154:Mutation; D009399:Nephrons / Q000002:abnormalities / Q000473:pathology / Q000648:ultrastructure; D009929:Organ Size; D009990:Oryzias / Q000002:abnormalities / Q000235:genetics*; D010641:Phenotype; D007690:Polycystic Kidney Diseases / Q000235:genetics* / Q000473:pathology*