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Gene Review

Rpe65  -  retinal pigment epithelium 65

Mus musculus

Synonyms: 65kDa, A930029L06Rik, All-trans-retinyl-palmitate hydrolase, LCA2, Mord1, ...
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Disease relevance of Rpe65


High impact information on Rpe65


Biological context of Rpe65


Anatomical context of Rpe65


Associations of Rpe65 with chemical compounds


Other interactions of Rpe65

  • We exposed to bright light two groups of genetically altered mice that lack the visual pigment rhodopsin (Rpe65-/- and Rho-/-) [4].
  • Despite the loss of rods and cones, the RPE65(-/-) and rd/rd maintained a 24-h circadian rhythm entrained to light similar to controls and were capable of circadian reentrainment to a 12-h light reversal [16].

Analytical, diagnostic and therapeutic context of Rpe65


  1. Lentiviral gene transfer of RPE65 rescues survival and function of cones in a mouse model of Leber congenital amaurosis. Bemelmans, A.P., Kostic, C., Crippa, S.V., Hauswirth, W.W., Lem, J., Munier, F.L., Seeliger, M.W., Wenzel, A., Arsenijevic, Y. PLoS Med. (2006) [Pubmed]
  2. Spontaneous activity of opsin apoprotein is a cause of Leber congenital amaurosis. Woodruff, M.L., Wang, Z., Chung, H.Y., Redmond, T.M., Fain, G.L., Lem, J. Nat. Genet. (2003) [Pubmed]
  3. Retinal degeneration and RPE transplantation in Rpe65(-/-) mice. Gouras, P., Kong, J., Tsang, S.H. Invest. Ophthalmol. Vis. Sci. (2002) [Pubmed]
  4. Protection of Rpe65-deficient mice identifies rhodopsin as a mediator of light-induced retinal degeneration. Grimm, C., Wenzel, A., Hafezi, F., Yu, S., Redmond, T.M., Remé, C.E. Nat. Genet. (2000) [Pubmed]
  5. Rpe65 is necessary for production of 11-cis-vitamin A in the retinal visual cycle. Redmond, T.M., Yu, S., Lee, E., Bok, D., Hamasaki, D., Chen, N., Goletz, P., Ma, J.X., Crouch, R.K., Pfeifer, K. Nat. Genet. (1998) [Pubmed]
  6. Noninvasive two-photon imaging reveals retinyl ester storage structures in the eye. Imanishi, Y., Batten, M.L., Piston, D.W., Baehr, W., Palczewski, K. J. Cell Biol. (2004) [Pubmed]
  7. Nonvisual light responses in the Rpe65 knockout mouse: rod loss restores sensitivity to the melanopsin system. Doyle, S.E., Castrucci, A.M., McCall, M., Provencio, I., Menaker, M. Proc. Natl. Acad. Sci. U.S.A. (2006) [Pubmed]
  8. Downregulation of cone-specific gene expression and degeneration of cone photoreceptors in the Rpe65-/- mouse at early ages. Znoiko, S.L., Rohrer, B., Lu, K., Lohr, H.R., Crouch, R.K., Ma, J.X. Invest. Ophthalmol. Vis. Sci. (2005) [Pubmed]
  9. Electroretinographic evidence for altered phototransduction gain and slowed recovery from photobleaches in albino mice with a MET450 variant in RPE65. Nusinowitz, S., Nguyen, L., Radu, R., Kashani, Z., Farber, D., Danciger, M. Exp. Eye Res. (2003) [Pubmed]
  10. The Rpe65 Leu450Met variation increases retinal resistance against light-induced degeneration by slowing rhodopsin regeneration. Wenzel, A., Reme, C.E., Williams, T.P., Hafezi, F., Grimm, C. J. Neurosci. (2001) [Pubmed]
  11. 11-cis-retinal reduces constitutive opsin phosphorylation and improves quantum catch in retinoid-deficient mouse rod photoreceptors. Ablonczy, Z., Crouch, R.K., Goletz, P.W., Redmond, T.M., Knapp, D.R., Ma, J.X., Rohrer, B. J. Biol. Chem. (2002) [Pubmed]
  12. Rpe65 is a retinyl ester binding protein that presents insoluble substrate to the isomerase in retinal pigment epithelial cells. Mata, N.L., Moghrabi, W.N., Lee, J.S., Bui, T.V., Radu, R.A., Horwitz, J., Travis, G.H. J. Biol. Chem. (2004) [Pubmed]
  13. Rpe65 Leu450Met variant is associated with reduced levels of the retinal pigment epithelium lipofuscin fluorophores A2E and iso-A2E. Kim, S.R., Fishkin, N., Kong, J., Nakanishi, K., Allikmets, R., Sparrow, J.R. Proc. Natl. Acad. Sci. U.S.A. (2004) [Pubmed]
  14. Retinyl esters are the substrate for isomerohydrolase. Moiseyev, G., Crouch, R.K., Goletz, P., Oatis, J., Redmond, T.M., Ma, J.X. Biochemistry (2003) [Pubmed]
  15. Opsin activation of transduction in the rods of dark-reared Rpe65 knockout mice. Fan, J., Woodruff, M.L., Cilluffo, M.C., Crouch, R.K., Fain, G.L. J. Physiol. (Lond.) (2005) [Pubmed]
  16. Entrainment of circadian rhythm to a photoperiod reversal shows retinal dystrophy in RPE65(-/-) mice. Daniels, D.M., Stoddart, C.W., Martin-Iverson, M.T., Lai, C.M., Redmond, T.M., Rakoczy, P.E. Physiol. Behav. (2003) [Pubmed]
  17. Light damage susceptibility and RPE65 in rats. Iseli, H.P., Wenzel, A., Hafezi, F., REme, C.E., Grimm, C. Exp. Eye Res. (2002) [Pubmed]
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