The retinol-binding protein receptor 2 (Rbpr2) is required for photoreceptor survival and visual function in the zebrafish

Glenn P. Lobo, Gayle Pauer, Joshua H. Lipschutz, Stephanie A. Hagstrom

Research output: Chapter in Book/Report/Conference proceedingChapter

Abstract

Vitamin A/retinol (ROL) and its metabolites (retinoids) play critical roles in eye development and photoreception. Short-term dietary vitamin A deficiency (VAD) manifests clinically as night blindness, while prolonged VAD is known to cause retinal pigment epithelium (RPE) and photoreceptor degeneration. Therefore, sustained uptake of dietary vitamin A, for ocular retinoid production, is essential for photoreceptor health and visual function. The mechanisms influencing the uptake, storage, and supply of dietary vitamin A, for ocular retinoid production, however, are not fully understood. We investigated, in zebrafish, the physiological role of the retinol-binding protein receptor 2 (Rbpr2), for the uptake of dietary ROL, which is necessary for vision. NIH3T3 cells expressing zebrafish Rbpr2 showed plasma membrane localization patterns and were capable of ROL uptake from its bound form. Using whole-mount in situ hybridization, Rbpr2 was found to be expressed exclusively in the liver, intestine, and pancreas, of staged zebrafish larvae. At 5.5 days post fertilization, TALEN-generated rbpr2 mutants (rbpr2 −/− ) had smaller eyes and shorter OS lengths and showed loss of PNA (cones) and rhodopsin (rods) by immunofluorescence staining. Finally, tests for visual function using optokinetic response (OKR) showed no consistent OKR in rbpr2 −/− larval zebrafish. Our analysis, therefore, suggests that Rbpr2 is capable of ROL uptake and loss of this membrane receptor in zebrafish results in photoreceptor defects that adversely affect visual function.

Original languageEnglish (US)
Title of host publicationAdvances in Experimental Medicine and Biology
PublisherSpringer New York LLC
Pages569-576
Number of pages8
DOIs
StatePublished - 2018
Externally publishedYes

Publication series

NameAdvances in Experimental Medicine and Biology
Volume1074
ISSN (Print)0065-2598
ISSN (Electronic)2214-8019

Bibliographical note

Funding Information:
Acknowledgments This study was supported by NIH grant EY025034-02 (GPL) and, in part, by a Knights Templar Eye Foundation Career-Starter Grant (GPL) and a Knights Templar Eye Foundation Career-Renewal Grant (GPL), NIH grant DK074038 (JHL), and a VA Merit award 2I01BX000820 (JHL).

Publisher Copyright:
© 2018, Springer International Publishing AG, part of Springer Nature.

Keywords

  • Photoreceptor
  • Rbpr2
  • Retinol
  • Vision
  • Vitamin A

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