title: Arrested differentiation and epithelial cell degeneration in zebrafish lens mutants.
abstract: In a chemical mutagenesis screen, we identified two zebrafish mutants that possessed small pupils. Genetic complementation revealed these two lines are due to mutations in different genes. The phenotypes of the two mutants were characterized using histologic, immunohistochemical, and tissue transplantation techniques. The arrested lens (arl) mutant exhibits a small eye and pupil phenotype at 48 hr postfertilization (hpf) and lacks any histologically identifiable lens structures by 5 days postfertilization (dpf). In contrast, the disrupted lens (dsl) mutants are phenotypically normal until 5 dpf, and then undergo lens disorganization and cell degeneration that is apparent by 7 dpf. Histology reveals the arl mutant terminates lens cell differentiation by 48 hpf, whereas the dsl lens exhibits a defective lens epithelial cell population at 5 dpf. Lens transplantation experiments demonstrate both mutations are autonomous to the lens tissue. Immunohistochemistry reveals the retinal cells may suffer subtle effects, possibly due to the lens abnormalities.
authors.affiliation: Center for Zebrafish Research and Department of Biological Sciences, University of Notre Dame, Notre Dame, Indiana 46556-0369, USA.
authors.forename: T S
authors.initials: TS
authors.lastname: Vihtelic
authors.forename: Y
authors.initials: Y
authors.lastname: Yamamoto
authors.forename: M T
authors.initials: MT
authors.lastname: Sweeney
authors.forename: W R
authors.initials: WR
authors.lastname: Jeffery
authors.forename: D R
authors.initials: DR
authors.lastname: Hyde
mesh_terms: D000818:Animals; D016923:Cell Death; D002454:Cell Differentiation; D004625:Embryo, Nonmammalian / Q000166:cytology / Q000378:metabolism; D004847:Epithelial Cells / Q000166:cytology* / Q000473:pathology*; D018398:Homeodomain Proteins / Q000378:metabolism; D007150:Immunohistochemistry; D007908:Lens, Crystalline / Q000196:embryology* / Q000637:transplantation; D008297:Male; D009154:Mutation / Q000502:physiology*; D010641:Phenotype; D012160:Retina / Q000166:cytology / Q000196:embryology; D025521:Tumor Suppressor Proteins; D015027:Zebrafish / Q000196:embryology*; D000098551:Prospero-Related Homeobox 1 Protein
chemical_list: D018398:Homeodomain Proteins; D025521:Tumor Suppressor Proteins; D000098551:Prospero-Related Homeobox 1 Protein