title: Wnt5a is necessary for normal kidney development in zebrafish and mice.
abstract: BACKGROUND
  Wnt5a is important for the development of various organs and postnatal cellular function. Little is known, however, about the role of Wnt5a in kidney development, although WNT5A mutations were identified in patients with Robinow syndrome, a genetic disease which includes developmental defects in kidneys. Our goal in this study was to determine the role of Wnt5a in kidney development.
  
  
  METHODS
  Whole-mount in situ hybridization was used to establish the expression pattern of Wnt5a during kidney development. Zebrafish with wnt5a knockdown and Wnt5a global knockout mice were used to identify kidney phenotypes.
  
  
  RESULTS
  In zebrafish, wnt5a knockdown resulted in glomerular cyst formation and dilated renal tubules. In mice, Wnt5a global knockout resulted in pleiotropic, but severe, kidney phenotypes, including agenesis, fused kidney, hydronephrosis and duplex kidney/ureter.
  
  
  CONCLUSIONS
  Our data demonstrated the important role of Wnt5a in kidney development. Disrupted Wnt5a resulted in kidney cysts in zebrafish and pleiotropic abnormal kidney development in mice.
authors.affiliation: Department of Medicine, Eastern Virginia Medical School, Norfolk, Va., USA.
authors.forename: Liwei
authors.initials: L
authors.lastname: Huang
authors.forename: An
authors.initials: A
authors.lastname: Xiao
authors.forename: Soo Young
authors.initials: SY
authors.lastname: Choi
authors.forename: Quane
authors.initials: Q
authors.lastname: Kan
authors.forename: Weibin
authors.initials: W
authors.lastname: Zhou
authors.forename: Maria F
authors.initials: MF
authors.lastname: Chacon-Heszele
authors.forename: Yun Kyoung
authors.initials: YK
authors.lastname: Ryu
authors.forename: Sarah
authors.initials: S
authors.lastname: McKenna
authors.forename: Xiaofeng
authors.initials: X
authors.lastname: Zuo
authors.forename: Rejji
authors.initials: R
authors.lastname: Kuruvilla
authors.forename: Joshua H
authors.initials: JH
authors.lastname: Lipschutz
mesh_terms: D000818:Animals; D004195:Disease Models, Animal; D005260:Female; D055786:Gene Knockout Techniques; D015994:Incidence; D007668:Kidney / Q000002:abnormalities / Q000196:embryology* / Q000502:physiology*; D052177:Kidney Diseases, Cystic / Q000453:epidemiology / Q000209:etiology / Q000503:physiopathology; D008297:Male; D051379:Mice; D018345:Mice, Knockout; D023421:Models, Animal; D051153:Wnt Proteins / Q000172:deficiency / Q000235:genetics / Q000502:physiology*; D000071818:Wnt-5a Protein; D015027:Zebrafish; D029961:Zebrafish Proteins / Q000172:deficiency / Q000235:genetics / Q000502:physiology*
chemical_list: D051153:Wnt Proteins; D000071818:Wnt-5a Protein; C438615:Wnt5a protein, mouse; C480282:Wnt5a protein, zebrafish; D029961:Zebrafish Proteins