title: Retinal defects in the zebrafish bleached mutant.
abstract: The recessive zebrafish mutant bleached has, apart from its defects in pigmentation, a heritable defect leading to larval blindness. Here, we analyze the retina of homozygous bleached larvae, employing morphological and electrophysiological methods. Electroretinography revealed a complete lack of electrical signals in response to light. Histological analysis of mutant retinae showed a severely affected outer retina with a hypopigmented pigment epithelium and a disorganized outer nuclear layer containing few or no intact photoreceptors. Using the TUNEL assay for cell death detection, we noticed a strong increase of apoptotic cells in all retinal cell layers, starting in young larvae even before retinal support of visual function. At later stages cell death is most pronounced at the marginal zone, where new cells are constantly added to the retina. At early stages increased apoptosis is mainly confined to the retina, while at later stages elevated cell death is al so apparent in extra-retinal tissues, particularly in the brain. Hence, the lack of visual responses in homozygous bleached larvae can be attributed to a severe defect of the outer retina, preceded by increased levels of apoptotic cell death in all retinal cell layers.
authors.affiliation: Brain Research Institute, University of Zurich and Department of Biology, Swiss Federal Institute of Technology Zurich, Winterthurerstr. 190, CH-8057 Zurich, Switzerland. neuhauss@hifo.unizh.ch
authors.forename: Stephan C F
authors.initials: SC
authors.lastname: Neuhauss
authors.forename: Mathias W
authors.initials: MW
authors.lastname: Seeliger
authors.forename: Carsten P
authors.initials: CP
authors.lastname: Schepp
authors.forename: Oliver
authors.initials: O
authors.lastname: Biehlmaier
mesh_terms: D000818:Animals; D017209:Apoptosis; D001766:Blindness / Q000209:etiology; D004596:Electroretinography; D005136:Eye Proteins / Q000235:genetics; D005808:Genes, Recessive; D007814:Larva; D009154:Mutation; D012160:Retina / Q000254:growth & development / Q000473:pathology / Q000503:physiopathology; D012162:Retinal Degeneration / Q000235:genetics* / Q000473:pathology / Q000503:physiopathology; D015027:Zebrafish / Q000235:genetics* / Q000254:growth & development; D029961:Zebrafish Proteins / Q000235:genetics*
chemical_list: D005136:Eye Proteins; D029961:Zebrafish Proteins