PUBLICATION

Evidence for RPE65-independent vision in the cone-dominated zebrafish retina

Authors
Schonthaler, H.B., Lampert, J.M., Isken, A., Rinner, O., Mader, A., Gesemann, M., Oberhauser, V., Golczak, M., Biehlmaier, O., Palczewski, K., Neuhauss, S.C., and von Lintig, J.
ID
ZDB-PUB-070920-15
Date
2007
Source
The European journal of neuroscience   26(7): 1940-1949 (Journal)
Registered Authors
Biehlmaier, Oliver, Lampert, Johanna, Neuhauss, Stephan, Rinner, Oliver
Keywords
none
MeSH Terms
  • Animals
  • Animals, Genetically Modified
  • Cell Line, Transformed
  • Diterpenes/pharmacology
  • Embryo, Nonmammalian
  • Gene Expression Regulation, Developmental/physiology*
  • Immunohistochemistry/methods
  • In Situ Hybridization/methods
  • Light
  • Mice
  • Retina/cytology*
  • Retina/enzymology*
  • Retinal Cone Photoreceptor Cells/physiology*
  • Retinaldehyde/metabolism
  • Vision, Ocular/physiology*
  • Zebrafish
  • cis-trans-Isomerases/genetics
  • cis-trans-Isomerases/physiology*
PubMed
17868371 Full text @ Eur. J. Neurosci.
CTD
17868371
Abstract
An enzyme-based cyclic pathway for trans to cis isomerization of the chromophore of visual pigments (11-cis-retinal) is intrinsic to vertebrate cone and rod vision. This process, called the visual cycle, is mostly characterized in rod-dominated retinas and essentially depends on RPE65, an all-trans to 11-cis-retinoid isomerase. Here we analysed the role of RPE65 in zebrafish, a species with a cone-dominated retina. We cloned zebrafish RPE65 and showed that its expression coincided with photoreceptor development. Targeted gene knockdown of RPE65 resulted in morphologically altered rod outer segments and overall reduced 11-cis-retinal levels. Cone vision of RPE65-deficient larvae remained functional as demonstrated by behavioural tests and by metabolite profiling for retinoids. Furthermore, all-trans retinylamine, a potent inhibitor of the rod visual cycle, reduced 11-cis-retinal levels of control larvae to a similar extent but showed no additive effects in RPE65-deficient larvae. Thus, our study of zebrafish provides in vivo evidence for the existence of an RPE65-independent pathway for the regeneration of 11-cis-retinal for cone vision.
Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping