PUBLICATION

The Retinol Binding Protein Receptor 2 (Rbpr2) is required for Photoreceptor Outer Segment Morphogenesis and Visual Function in Zebrafish

Authors
Shi, Y., Obert, E., Rahman, B., Rohrer, B., Lobo, G.P.
ID
ZDB-PUB-171128-15
Date
2017
Source
Scientific Reports   7: 16207 (Journal)
Registered Authors
Lobo, Glenn, Shi, Yi
Keywords
none
MeSH Terms
  • Vision, Ocular
  • Zebrafish
  • Gene Expression Regulation, Developmental
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
  • Enterocytes/metabolism
  • Hepatocytes/metabolism
  • Retinal Photoreceptor Cell Outer Segment/metabolism*
  • Retinal Photoreceptor Cell Outer Segment/physiology
  • Animals
  • Morphogenesis*
  • Membrane Transport Proteins/genetics
  • Membrane Transport Proteins/metabolism*
(all 13)
PubMed
29176573 Full text @ Sci. Rep.
Abstract
Vitamin A (all-trans retinol) plays critical roles in mammalian development and vision. Since vitamin A is food-derived, tissue-specific uptake and storage mechanism are needed. In the eye, uptake of RBP4-retinol is mediated by the receptor Stra6, whereas the receptor mediating RBP4 binding and retinol transport into the liver has just recently been discovered. Here we examined the role of zebrafish retinol binding protein receptor 2 (Rbpr2) for RBP4-retinol uptake in developing embryos, using eye development and vision as sensitive readouts. In cultured cells, Rbpr2 localized to membranes and promoted RBP4-retinol uptake. In larvae, Rbpr2 expression was detected in developing intestinal enterocytes and liver hepatocytes. Two rbpr2 mutant zebrafish lines, each resulting in Rbpr2 deficiency, exhibit a small eye defect, and systemic malformations including hydrocephaly and cardiac edema, phenotypes associated with vitamin A deficiency. In the retina, Rbpr2 loss resulted in shorter photoreceptor outer segments, mislocalization and decrease in visual pigments, decreased expression of retinoic acid-responsive genes and photoreceptor cell loss, overall leading to a reduction of visual function. Together, these results demonstrate that Rbpr2-mediated RBP4-retinol uptake in developing liver and intestine is necessary to provide sufficient substrate for ocular retinoid production required for photoreceptor cell maintenance and visual function.
Genes / Markers
Figures
Figure Gallery (7 images)
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
muz97
    Insertion
    sa10706
      Point Mutation
      1 - 2 of 2
      Show
      Human Disease / Model
      No data available
      Sequence Targeting Reagents
      Target Reagent Reagent Type
      stra6lTALEN1-stra6lTALEN
      1 - 1 of 1
      Show
      Fish
      Antibodies
      Name Type Antigen Genes Isotypes Host Organism
      Ab1-opn1lwpolyclonal
        Rabbit
        Ab3-rhomonoclonalIgG1Mouse
        1 - 2 of 2
        Show
        Orthology
        No data available
        Engineered Foreign Genes
        No data available
        Mapping
        No data available