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Utility of In Vitro Mutagenesis of RPE65 Protein for Verification of Mutational Pathogenicity Before Gene Therapy.

Yang U, Gentleman S, Gai X, Gorin MB, Borchert MS, Lee TC, Villanueva A, Koenekoop R, Maguire AM, Bennett J, Redmond TM, Nagiel A.

JAMA Ophthalmol. 2019 Oct 3:1-9. doi: 10.1001/jamaophthalmol.2019.3914. [Epub ahead of print]


Stable Intronic Sequences and Exon Skipping Events in the Human RPE65 Gene: Analysis of Expression in Retinal Pigment Epithelium Cells and Cell Culture Models.

Postnikova O, Poliakov E, Golestaneh N, Rogozin IB, Redmond TM.

Front Genet. 2019 Jul 19;10:634. doi: 10.3389/fgene.2019.00634. eCollection 2019.


The dual roles of RPE65 S-palmitoylation in membrane association and visual cycle function.

Uppal S, Liu T, Poliakov E, Gentleman S, Redmond TM.

Sci Rep. 2019 Mar 26;9(1):5218. doi: 10.1038/s41598-019-41501-w.


Volatile Evolution of Long Non-Coding RNA Repertoire in Retinal Pigment Epithelium: Insights from Comparison of Bovine and Human RNA Expression Profiles.

Postnikova OA, Rogozin IB, Samuel W, Nudelman G, Babenko VN, Poliakov E, Redmond TM.

Genes (Basel). 2019 Mar 8;10(3). pii: E205. doi: 10.3390/genes10030205.


Aberrant RNA splicing is the major pathogenic effect in a knock-in mouse model of the dominantly inherited c.1430A>G human RPE65 mutation.

Li Y, Furhang R, Ray A, Duncan T, Soucy J, Mahdi R, Chaitankar V, Gieser L, Poliakov E, Qian H, Liu P, Dong L, Rogozin IB, Redmond TM.

Hum Mutat. 2019 Apr;40(4):426-443. doi: 10.1002/humu.23706. Epub 2019 Jan 25.


Expression of ABCA4 in the retinal pigment epithelium and its implications for Stargardt macular degeneration.

Lenis TL, Hu J, Ng SY, Jiang Z, Sarfare S, Lloyd MB, Esposito NJ, Samuel W, Jaworski C, Bok D, Finnemann SC, Radeke MJ, Redmond TM, Travis GH, Radu RA.

Proc Natl Acad Sci U S A. 2018 Nov 20;115(47):E11120-E11127. doi: 10.1073/pnas.1802519115. Epub 2018 Nov 5.


Proinflammatory cytokine interferon-γ increases the expression of BANCR, a long non-coding RNA, in retinal pigment epithelial cells.

Kutty RK, Samuel W, Duncan T, Postnikova O, Jaworski C, Nagineni CN, Redmond TM.

Cytokine. 2018 Apr;104:147-150. doi: 10.1016/j.cyto.2017.10.009. Epub 2017 Oct 18.


Phylogenetic analysis of the metazoan carotenoid oxygenase superfamily: a new ancestral gene assemblage of BCO-like (BCOL) proteins.

Poliakov E, Soucy J, Gentleman S, Rogozin IB, Redmond TM.

Sci Rep. 2017 Oct 16;7(1):13192. doi: 10.1038/s41598-017-13521-x.


RPE65 takes on another role in the vertebrate retina.

Redmond TM.

Proc Natl Acad Sci U S A. 2017 Oct 10;114(41):10818-10820. doi: 10.1073/pnas.1715064114. Epub 2017 Oct 2. No abstract available.


Inhibition of thyroid hormone receptor locally in the retina is a therapeutic strategy for retinal degeneration.

Ma H, Yang F, Butler MR, Belcher J, Redmond TM, Placzek AT, Scanlan TS, Ding XQ.

FASEB J. 2017 Aug;31(8):3425-3438. doi: 10.1096/fj.201601166RR. Epub 2017 Apr 20.


Appropriately differentiated ARPE-19 cells regain phenotype and gene expression profiles similar to those of native RPE cells.

Samuel W, Jaworski C, Postnikova OA, Kutty RK, Duncan T, Tan LX, Poliakov E, Lakkaraju A, Redmond TM.

Mol Vis. 2017 Mar 5;23:60-89. eCollection 2017.


Proinflammatory cytokines decrease the expression of genes critical for RPE function.

Kutty RK, Samuel W, Boyce K, Cherukuri A, Duncan T, Jaworski C, Nagineni CN, Redmond TM.

Mol Vis. 2016 Oct 8;22:1156-1168. eCollection 2016.


Targeting iodothyronine deiodinases locally in the retina is a therapeutic strategy for retinal degeneration.

Yang F, Ma H, Belcher J, Butler MR, Redmond TM, Boye SL, Hauswirth WW, Ding XQ.

FASEB J. 2016 Dec;30(12):4313-4325. Epub 2016 Sep 13.


Inhibition of RPE65 Retinol Isomerase Activity by Inhibitors of Lipid Metabolism.

Eroglu A, Gentleman S, Poliakov E, Redmond TM.

J Biol Chem. 2016 Mar 4;291(10):4966-73. doi: 10.1074/jbc.M115.685651. Epub 2015 Dec 30.


A History of the Classical Visual Cycle.

Wright CB, Redmond TM, Nickerson JM.

Prog Mol Biol Transl Sci. 2015;134:433-48. doi: 10.1016/bs.pmbts.2015.06.009. Epub 2015 Jul 3. Review.


Mouse model of human RPE65 P25L hypomorph resembles wild type under normal light rearing but is fully resistant to acute light damage.

Li Y, Yu S, Duncan T, Li Y, Liu P, Gene E, Cortes-Pena Y, Qian H, Dong L, Redmond TM.

Hum Mol Genet. 2015 Aug 1;24(15):4417-28. doi: 10.1093/hmg/ddv178. Epub 2015 May 13.


Resveratrol attenuates CXCL11 expression induced by proinflammatory cytokines in retinal pigment epithelial cells.

Kutty RK, Samuel W, Abay R, Cherukuri A, Nagineni CN, Duncan T, Jaworski C, Vijayasarathy C, Redmond TM.

Cytokine. 2015 Aug;74(2):335-8. doi: 10.1016/j.cyto.2015.03.016. Epub 2015 Apr 15.


Transcriptional regulatory events initiated by Ascl1 and Neurog2 during neuronal differentiation of P19 embryonic carcinoma cells.

Huang HS, Redmond TM, Kubish GM, Gupta S, Thompson RC, Turner DL, Uhler MD.

J Mol Neurosci. 2015 Mar;55(3):684-705. doi: 10.1007/s12031-014-0408-2. Epub 2014 Sep 6.


Multiple A2E treatments lead to melanization of rod outer segment-challenged ARPE-19 cells.

Poliakov E, Strunnikova NV, Jiang JK, Martinez B, Parikh T, Lakkaraju A, Thomas C, Brooks BP, Redmond TM.

Mol Vis. 2014 Mar 14;20:285-300. eCollection 2014.


The Rpe65 rd12 allele exerts a semidominant negative effect on vision in mice.

Wright CB, Chrenek MA, Feng W, Getz SE, Duncan T, Pardue MT, Feng Y, Redmond TM, Boatright JH, Nickerson JM.

Invest Ophthalmol Vis Sci. 2014 Apr 17;55(4):2500-15. doi: 10.1167/iovs.13-13574.


Suppressing thyroid hormone signaling preserves cone photoreceptors in mouse models of retinal degeneration.

Ma H, Thapa A, Morris L, Redmond TM, Baehr W, Ding XQ.

Proc Natl Acad Sci U S A. 2014 Mar 4;111(9):3602-7. doi: 10.1073/pnas.1317041111. Epub 2014 Feb 18.


Fenretinide induces ubiquitin-dependent proteasomal degradation of stearoyl-CoA desaturase in human retinal pigment epithelial cells.

Samuel W, Kutty RK, Duncan T, Vijayasarathy C, Kuo BC, Chapa KM, Redmond TM.

J Cell Physiol. 2014 Aug;229(8):1028-38. doi: 10.1002/jcp.24527.


Complementation test of Rpe65 knockout and tvrm148.

Wright CB, Chrenek MA, Foster SL, Duncan T, Redmond TM, Pardue MT, Boatright JH, Nickerson JM.

Invest Ophthalmol Vis Sci. 2013 Jul 30;54(7):5111-22. doi: 10.1167/iovs.13-12336.


Differential regulation of microRNA-146a and microRNA-146b-5p in human retinal pigment epithelial cells by interleukin-1β, tumor necrosis factor-α, and interferon-γ.

Kutty RK, Nagineni CN, Samuel W, Vijayasarathy C, Jaworski C, Duncan T, Cameron JE, Flemington EK, Hooks JJ, Redmond TM.

Mol Vis. 2013 Apr 3;19:737-50. Print 2013.


Origin and evolution of retinoid isomerization machinery in vertebrate visual cycle: hint from jawless vertebrates.

Poliakov E, Gubin AN, Stearn O, Li Y, Campos MM, Gentleman S, Rogozin IB, Redmond TM.

PLoS One. 2012;7(11):e49975. doi: 10.1371/journal.pone.0049975. Epub 2012 Nov 27.


Aromatic residues in the substrate cleft of RPE65 protein govern retinol isomerization and modulate its progression.

Chander P, Gentleman S, Poliakov E, Redmond TM.

J Biol Chem. 2012 Aug 31;287(36):30552-9. doi: 10.1074/jbc.M112.364596. Epub 2012 Jun 28.


Subretinal delivery and electroporation in pigmented and nonpigmented adult mouse eyes.

Nickerson JM, Goodman P, Chrenek MA, Bernal CJ, Berglin L, Redmond TM, Boatright JH.

Methods Mol Biol. 2012;884:53-69. doi: 10.1007/978-1-61779-848-1_4.


In vivo assessment of rodent retinal structure using spectral domain optical coherence tomography.

Fischer MD, Huber G, Paquet-Durand F, Humphries P, Redmond TM, Grimm C, Seeliger MW.

Adv Exp Med Biol. 2012;723:489-94. doi: 10.1007/978-1-4614-0631-0_61. No abstract available.


The mechanism of fenretinide (4-HPR) inhibition of β-carotene monooxygenase 1. New suspect for the visual side effects of fenretinide.

Poliakov E, Gubin A, Laird J, Gentleman S, Salomon RG, Redmond TM.

Adv Exp Med Biol. 2012;723:167-74. doi: 10.1007/978-1-4614-0631-0_23. No abstract available.


Aromatic lipophilic spin traps effectively inhibit RPE65 isomerohydrolase activity.

Poliakov E, Parikh T, Ayele M, Kuo S, Chander P, Gentleman S, Redmond TM.

Biochemistry. 2011 Aug 16;50(32):6739-41. doi: 10.1021/bi200532m. Epub 2011 Jul 13.


A novel fluorescent transcriptional reporter for cell-based microarray assays.

Redmond TM, Uhler MD.

Methods Mol Biol. 2011;706:41-52. doi: 10.1007/978-1-61737-970-3_4.


Inflammatory cytokines regulate microRNA-155 expression in human retinal pigment epithelial cells by activating JAK/STAT pathway.

Kutty RK, Nagineni CN, Samuel W, Vijayasarathy C, Hooks JJ, Redmond TM.

Biochem Biophys Res Commun. 2010 Nov 12;402(2):390-5. doi: 10.1016/j.bbrc.2010.10.042. Epub 2010 Oct 13.


MicroRNA expression in human retinal pigment epithelial (ARPE-19) cells: increased expression of microRNA-9 by N-(4-hydroxyphenyl)retinamide.

Kutty RK, Samuel W, Jaworski C, Duncan T, Nagineni CN, Raghavachari N, Wiggert B, Redmond TM.

Mol Vis. 2010 Aug 4;16:1475-86.


Direct transcriptional induction of Gadd45gamma by Ascl1 during neuronal differentiation.

Huang HS, Kubish GM, Redmond TM, Turner DL, Thompson RC, Murphy GG, Uhler MD.

Mol Cell Neurosci. 2010 Jul;44(3):282-96. doi: 10.1016/j.mcn.2010.03.014. Epub 2010 Apr 9.


Biochemical evidence for the tyrosine involvement in cationic intermediate stabilization in mouse beta-carotene 15, 15'-monooxygenase.

Poliakov E, Gentleman S, Chander P, Cunningham FX Jr, Grigorenko BL, Nemuhin AV, Redmond TM.

BMC Biochem. 2009 Dec 14;10:31. doi: 10.1186/1471-2091-10-31.


Gene therapy for Leber's congenital amaurosis is safe and effective through 1.5 years after vector administration.

Simonelli F, Maguire AM, Testa F, Pierce EA, Mingozzi F, Bennicelli JL, Rossi S, Marshall K, Banfi S, Surace EM, Sun J, Redmond TM, Zhu X, Shindler KS, Ying GS, Ziviello C, Acerra C, Wright JF, McDonnell JW, High KA, Bennett J, Auricchio A.

Mol Ther. 2010 Mar;18(3):643-50. doi: 10.1038/mt.2009.277. Epub 2009 Dec 1.


RPE65, visual cycle retinol isomerase, is not inherently 11-cis-specific: support for a carbocation mechanism of retinol isomerization.

Redmond TM, Poliakov E, Kuo S, Chander P, Gentleman S.

J Biol Chem. 2010 Jan 15;285(3):1919-27. doi: 10.1074/jbc.M109.027458. Epub 2009 Nov 17.


Age-dependent effects of RPE65 gene therapy for Leber's congenital amaurosis: a phase 1 dose-escalation trial.

Maguire AM, High KA, Auricchio A, Wright JF, Pierce EA, Testa F, Mingozzi F, Bennicelli JL, Ying GS, Rossi S, Fulton A, Marshall KA, Banfi S, Chung DC, Morgan JI, Hauck B, Zelenaia O, Zhu X, Raffini L, Coppieters F, De Baere E, Shindler KS, Volpe NJ, Surace EM, Acerra C, Lyubarsky A, Redmond TM, Stone E, Sun J, McDonnell JW, Leroy BP, Simonelli F, Bennett J.

Lancet. 2009 Nov 7;374(9701):1597-605. doi: 10.1016/S0140-6736(09)61836-5. Epub 2009 Oct 23. Erratum in: Lancet. 2010 Jan 2;375(9708):30.


Spectral domain optical coherence tomography in mouse models of retinal degeneration.

Huber G, Beck SC, Grimm C, Sahaboglu-Tekgoz A, Paquet-Durand F, Wenzel A, Humphries P, Redmond TM, Seeliger MW, Fischer MD.

Invest Ophthalmol Vis Sci. 2009 Dec;50(12):5888-95. doi: 10.1167/iovs.09-3724. Epub 2009 Aug 6.


Technical brief: subretinal injection and electroporation into adult mouse eyes.

Johnson CJ, Berglin L, Chrenek MA, Redmond TM, Boatright JH, Nickerson JM.

Mol Vis. 2008;14:2211-26. Epub 2008 Dec 5.


Focus on Molecules: RPE65, the visual cycle retinol isomerase.

Redmond TM.

Exp Eye Res. 2009 May;88(5):846-7. doi: 10.1016/j.exer.2008.07.015. Epub 2008 Aug 14. Review. No abstract available.


A comprehensive clinical and biochemical functional study of a novel RPE65 hypomorphic mutation.

Lorenz B, Poliakov E, Schambeck M, Friedburg C, Preising MN, Redmond TM.

Invest Ophthalmol Vis Sci. 2008 Dec;49(12):5235-42. doi: 10.1167/iovs.07-1671. Epub 2008 Jul 3.


Safety and efficacy of gene transfer for Leber's congenital amaurosis.

Maguire AM, Simonelli F, Pierce EA, Pugh EN Jr, Mingozzi F, Bennicelli J, Banfi S, Marshall KA, Testa F, Surace EM, Rossi S, Lyubarsky A, Arruda VR, Konkle B, Stone E, Sun J, Jacobs J, Dell'Osso L, Hertle R, Ma JX, Redmond TM, Zhu X, Hauck B, Zelenaia O, Shindler KS, Maguire MG, Wright JF, Volpe NJ, McDonnell JW, Auricchio A, High KA, Bennett J.

N Engl J Med. 2008 May 22;358(21):2240-8. doi: 10.1056/NEJMoa0802315. Epub 2008 Apr 27.


Mitogen-activated protein kinase pathway mediates N-(4-hydroxyphenyl)retinamide-induced neuronal differentiation in the ARPE-19 human retinal pigment epithelial cell line.

Samuel W, Kutty RK, Sekhar S, Vijayasarathy C, Wiggert B, Redmond TM.

J Neurochem. 2008 Jul;106(2):591-602. doi: 10.1111/j.1471-4159.2008.05409.x. Epub 2008 Apr 10.


Assessing the efficacy of gene therapy in Rpe65-/- mice using photoentrainment of circadian rhythm.

Stoddart CW, Yu MJ, Martin-Iverson MT, Daniels DM, Lai CM, Barnett NL, Redmond TM, Narfström K, Rakoczyt PE.

Adv Exp Med Biol. 2006;572:239-45. No abstract available.


Identification of a KRAB-zinc finger protein binding to the Rpe65 gene promoter.

Lu Z, Poliakov E, Redmond TM.

Curr Eye Res. 2006 May;31(5):457-66.


9-cis Retinal increased in retina of RPE65 knockout mice with decrease in coat pigmentation.

Fan J, Wu BX, Sarna T, Rohrer B, Redmond TM, Crouch RK.

Photochem Photobiol. 2006 Nov-Dec;82(6):1461-7.


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