We study epithelial organ development and regeneration using the zebrafish as a model system. We established the zebrafish pronephros as a model for studying kidney development, cilia and ciliopathies, human kidney disease pathology, and kidney injury/regeneration. We study how the kidney develops, identify zebrafish genetic mutants, and determine key points of equivalence between fish and mammalian kidneys that make zebrafish a relevant model of human disease. Zebrafish provide a unique window on organogenesis that enables discovery of novel and broadly conserved developmental and regenerative mechanisms.
The zebrafish plays an increasingly important role in biomedical genetics because they facilitate discovery of basic developmental and regenerative mechanisms, provide an unbiased route to disease gene discovery, and enable development of in vivo assays to determine the significance of sequence variants found in human DNA sequence. Zebrafish embryo transparency also allows for live, in vivo cell imaging to follow morphogenesis and cell signaling at single cell resolution.
Our initial publication described zebrafish pronephric kidney development and cell structure, defined assays for nephron function, and characterized fifteen ENU mutant loci that caused kidney cyst formation and altered epithelial membrane protein targeting.