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Growth hormone regulates opsin expression in the retina of a salmonid fish.

Novales Flamarique I, Sayed Ahmed A, Cheng CL, Molday RS, Devlin RH.

J Neuroendocrinol. 2019 Nov;31(11):e12804. doi: 10.1111/jne.12804. Epub 2019 Nov 13.


ATP8A2-related disorders as recessive cerebellar ataxia.

Guissart C, Harrison AN, Benkirane M, Oncel I, Arslan EA, Chassevent AK, Baraῆano K, Larrieu L, Iascone M, Tenconi R, Claustres M, Eroglu-Ertugrul N, Calvas P, Topaloglu H, Molday RS, Koenig M.

J Neurol. 2020 Jan;267(1):203-213. doi: 10.1007/s00415-019-09579-4. Epub 2019 Oct 14.


Dual ABCA4-AAV Vector Treatment Reduces Pathogenic Retinal A2E Accumulation in a Mouse Model of Autosomal Recessive Stargardt Disease.

Dyka FM, Molday LL, Chiodo VA, Molday RS, Hauswirth WW.

Hum Gene Ther. 2019 Nov;30(11):1361-1370. doi: 10.1089/hum.2019.132. Epub 2019 Sep 30.


Expression and functional characterization of missense mutations in ATP8A2 linked to severe neurological disorders.

Choi H, Andersen JP, Molday RS.

Hum Mutat. 2019 Dec;40(12):2353-2364. doi: 10.1002/humu.23889. Epub 2019 Aug 23.


Phosphatidylserine flipping by the P4-ATPase ATP8A2 is electrogenic.

Tadini-Buoninsegni F, Mikkelsen SA, Mogensen LS, Molday RS, Andersen JP.

Proc Natl Acad Sci U S A. 2019 Aug 13;116(33):16332-16337. doi: 10.1073/pnas.1910211116. Epub 2019 Aug 1.


Identification and functional analyses of disease-associated P4-ATPase phospholipid flippase variants in red blood cells.

Liou AY, Molday LL, Wang J, Andersen JP, Molday RS.

J Biol Chem. 2019 Apr 26;294(17):6809-6821. doi: 10.1074/jbc.RA118.007270. Epub 2019 Mar 8.


Asparagine 905 of the mammalian phospholipid flippase ATP8A2 is essential for lipid substrate-induced activation of ATP8A2 dephosphorylation.

Mikkelsen SA, Mogensen LS, Vilsen B, Molday RS, Vestergaard AL, Andersen JP.

J Biol Chem. 2019 Apr 12;294(15):5970-5979. doi: 10.1074/jbc.RA118.007240. Epub 2019 Feb 13.


Cell-Specific Markers for the Identification of Retinal Cells and Subcellular Organelles by Immunofluorescence Microscopy.

Molday LL, Cheng CL, Molday RS.

Methods Mol Biol. 2019;1834:293-310. doi: 10.1007/978-1-4939-8669-9_19.


Proteomic Analysis and Functional Characterization of P4-ATPase Phospholipid Flippases from Murine Tissues.

Wang J, Molday LL, Hii T, Coleman JA, Wen T, Andersen JP, Molday RS.

Sci Rep. 2018 Jul 17;8(1):10795. doi: 10.1038/s41598-018-29108-z.


Recessive mutations in ATP8A2 cause severe hypotonia, cognitive impairment, hyperkinetic movement disorders and progressive optic atrophy.

McMillan HJ, Telegrafi A, Singleton A, Cho MT, Lelli D, Lynn FC, Griffin J, Asamoah A, Rinne T, Erasmus CE, Koolen DA, Haaxma CA, Keren B, Doummar D, Mignot C, Thompson I, Velsher L, Dehghani M, Vahidi Mehrjardi MY, Maroofian R, Tchan M, Simons C, Christodoulou J, Martín-Hernández E, Guillen Sacoto MJ, Henderson LB, McLaughlin H, Molday LL, Molday RS, Yoon G.

Orphanet J Rare Dis. 2018 May 31;13(1):86. doi: 10.1186/s13023-018-0825-3.


Correlating the Expression and Functional Activity of ABCA4 Disease Variants With the Phenotype of Patients With Stargardt Disease.

Garces F, Jiang K, Molday LL, Stöhr H, Weber BH, Lyons CJ, Maberley D, Molday RS.

Invest Ophthalmol Vis Sci. 2018 May 1;59(6):2305-2315. doi: 10.1167/iovs.17-23364.


Ca2+-activated Cl current predominates in threshold response of mouse olfactory receptor neurons.

Li RC, Lin CC, Ren X, Wu JS, Molday LL, Molday RS, Yau KW.

Proc Natl Acad Sci U S A. 2018 May 22;115(21):5570-5575. doi: 10.1073/pnas.1803443115. Epub 2018 May 7.


Disease mutations reveal residues critical to the interaction of P4-ATPases with lipid substrates.

Gantzel RH, Mogensen LS, Mikkelsen SA, Vilsen B, Molday RS, Vestergaard AL, Andersen JP.

Sci Rep. 2017 Sep 5;7(1):10418. doi: 10.1038/s41598-017-10741-z.


Peripherin diverts ciliary ectosome release to photoreceptor disc morphogenesis.

Molday RS, Goldberg AFX.

J Cell Biol. 2017 May 1;216(5):1227-1229. doi: 10.1083/jcb.201703020. Epub 2017 Apr 11.


C-terminus of the P4-ATPase ATP8A2 functions in protein folding and regulation of phospholipid flippase activity.

Chalat M, Moleschi K, Molday RS.

Mol Biol Cell. 2017 Feb 1;28(3):452-462. doi: 10.1091/mbc.E16-06-0453. Epub 2016 Dec 8.


PAX6 MiniPromoters drive restricted expression from rAAV in the adult mouse retina.

Hickmott JW, Chen CY, Arenillas DJ, Korecki AJ, Lam SL, Molday LL, Bonaguro RJ, Zhou M, Chou AY, Mathelier A, Boye SL, Hauswirth WW, Molday RS, Wasserman WW, Simpson EM.

Mol Ther Methods Clin Dev. 2016 Aug 10;3:16051. doi: 10.1038/mtm.2016.51. eCollection 2016.


P4-ATPases as Phospholipid Flippases-Structure, Function, and Enigmas.

Andersen JP, Vestergaard AL, Mikkelsen SA, Mogensen LS, Chalat M, Molday RS.

Front Physiol. 2016 Jul 8;7:275. doi: 10.3389/fphys.2016.00275. eCollection 2016. Review.


Cog-Wheel Octameric Structure of RS1, the Discoidin Domain Containing Retinal Protein Associated with X-Linked Retinoschisis.

Bush M, Setiaputra D, Yip CK, Molday RS.

PLoS One. 2016 Jan 26;11(1):e0147653. doi: 10.1371/journal.pone.0147653. eCollection 2016.


Specific mutations in mammalian P4-ATPase ATP8A2 catalytic subunit entail differential glycosylation of the accessory CDC50A subunit.

Vestergaard AL, Mikkelsen SA, Coleman JA, Molday RS, Vilsen B, Andersen JP.

FEBS Lett. 2015 Dec 21;589(24 Pt B):3908-14. doi: 10.1016/j.febslet.2015.11.031. Epub 2015 Nov 22.


Photoreceptors at a glance.

Molday RS, Moritz OL.

J Cell Sci. 2015 Nov 15;128(22):4039-45. doi: 10.1242/jcs.175687. Review.


Insights into the Molecular Properties of ABCA4 and Its Role in the Visual Cycle and Stargardt Disease.

Molday RS.

Prog Mol Biol Transl Sci. 2015;134:415-31. doi: 10.1016/bs.pmbts.2015.06.008. Epub 2015 Jul 14. Review.


A new mouse model for stationary night blindness with mutant Slc24a1 explains the pathophysiology of the associated human disease.

Vinberg F, Wang T, Molday RS, Chen J, Kefalov VJ.

Hum Mol Genet. 2015 Oct 15;24(20):5915-29. doi: 10.1093/hmg/ddv319. Epub 2015 Aug 5.


Segregating phototransduction from morphogenesis in photoreceptor outer segments.

Molday RS.

Channels (Austin). 2015;9(2):59-60. doi: 10.1080/19336950.2015.1017999. No abstract available.


Advancing therapeutic strategies for inherited retinal degeneration: recommendations from the Monaciano Symposium.

Thompson DA, Ali RR, Banin E, Branham KE, Flannery JG, Gamm DM, Hauswirth WW, Heckenlively JR, Iannaccone A, Jayasundera KT, Khan NW, Molday RS, Pennesi ME, Reh TA, Weleber RG, Zacks DN; Monaciano Consortium.

Invest Ophthalmol Vis Sci. 2015 Feb 9;56(2):918-31. doi: 10.1167/iovs.14-16049. Review.


Transport through recycling endosomes requires EHD1 recruitment by a phosphatidylserine translocase.

Lee S, Uchida Y, Wang J, Matsudaira T, Nakagawa T, Kishimoto T, Mukai K, Inaba T, Kobayashi T, Molday RS, Taguchi T, Arai H.

EMBO J. 2015 Mar 4;34(5):669-88. doi: 10.15252/embj.201489703. Epub 2015 Jan 16.


Impaired association of retinal degeneration-3 with guanylate cyclase-1 and guanylate cyclase-activating protein-1 leads to leber congenital amaurosis-1.

Zulliger R, Naash MI, Rajala RV, Molday RS, Azadi S.

J Biol Chem. 2015 Feb 6;290(6):3488-99. doi: 10.1074/jbc.M114.616656. Epub 2014 Dec 4.


A combined transgenic proteomic analysis and regulated trafficking of neuroligin-2.

Kang Y, Ge Y, Cassidy RM, Lam V, Luo L, Moon KM, Lewis R, Molday RS, Wong RO, Foster LJ, Craig AM.

J Biol Chem. 2014 Oct 17;289(42):29350-64. doi: 10.1074/jbc.M114.549279. Epub 2014 Sep 4.


1D4: a versatile epitope tag for the purification and characterization of expressed membrane and soluble proteins.

Molday LL, Molday RS.

Methods Mol Biol. 2014;1177:1-15. doi: 10.1007/978-1-4939-1034-2_1.


Insights into the role of RD3 in guanylate cyclase trafficking, photoreceptor degeneration, and Leber congenital amaurosis.

Molday LL, Jefferies T, Molday RS.

Front Mol Neurosci. 2014 May 26;7:44. doi: 10.3389/fnmol.2014.00044. eCollection 2014. Review.


ATP-binding cassette transporter ABCA4 and chemical isomerization protect photoreceptor cells from the toxic accumulation of excess 11-cis-retinal.

Quazi F, Molday RS.

Proc Natl Acad Sci U S A. 2014 Apr 1;111(13):5024-9. doi: 10.1073/pnas.1400780111. Epub 2014 Mar 20.


Critical roles of isoleucine-364 and adjacent residues in a hydrophobic gate control of phospholipid transport by the mammalian P4-ATPase ATP8A2.

Vestergaard AL, Coleman JA, Lemmin T, Mikkelsen SA, Molday LL, Vilsen B, Molday RS, Dal Peraro M, Andersen JP.

Proc Natl Acad Sci U S A. 2014 Apr 8;111(14):E1334-43. doi: 10.1073/pnas.1321165111. Epub 2014 Mar 24. Erratum in: Proc Natl Acad Sci U S A. 2014 Nov 18;111(46):16629.


Phospholipid flippase ATP8A2 is required for normal visual and auditory function and photoreceptor and spiral ganglion cell survival.

Coleman JA, Zhu X, Djajadi HR, Molday LL, Smith RS, Libby RT, John SW, Molday RS.

J Cell Sci. 2014 Mar 1;127(Pt 5):1138-49. doi: 10.1242/jcs.145052. Epub 2014 Jan 10.


Interaction of 4.1G and cGMP-gated channels in rod photoreceptor outer segments.

Cheng CL, Molday RS.

J Cell Sci. 2013 Dec 15;126(Pt 24):5725-34. doi: 10.1242/jcs.137679. Epub 2013 Oct 21.


Differential phospholipid substrates and directional transport by ATP-binding cassette proteins ABCA1, ABCA7, and ABCA4 and disease-causing mutants.

Quazi F, Molday RS.

J Biol Chem. 2013 Nov 29;288(48):34414-26. doi: 10.1074/jbc.M113.508812. Epub 2013 Oct 4.


RD3 gene delivery restores guanylate cyclase localization and rescues photoreceptors in the Rd3 mouse model of Leber congenital amaurosis 12.

Molday LL, Djajadi H, Yan P, Szczygiel L, Boye SL, Chiodo VA, Gregory-Evans K, Sarunic MV, Hauswirth WW, Molday RS.

Hum Mol Genet. 2013 Oct 1;22(19):3894-905. doi: 10.1093/hmg/ddt244. Epub 2013 Jun 4.


Changes in gene expression associated with retinal degeneration in the rd3 mouse.

Cheng CL, Molday RS.

Mol Vis. 2013 May 6;19:955-69. Print 2013.


Molecular organization and ATP-induced conformational changes of ABCA4, the photoreceptor-specific ABC transporter.

Tsybovsky Y, Orban T, Molday RS, Taylor D, Palczewski K.

Structure. 2013 May 7;21(5):854-60. doi: 10.1016/j.str.2013.03.001. Epub 2013 Apr 4.


Cholesterol transport via ABCA1: new insights from solid-phase binding assay.

Reboul E, Dyka FM, Quazi F, Molday RS.

Biochimie. 2013 Apr;95(4):957-61. doi: 10.1016/j.biochi.2012.11.009. Epub 2012 Nov 27.


Cell-specific markers for the identification of retinal cells by immunofluorescence microscopy.

Cheng CL, Djajadi H, Molday RS.

Methods Mol Biol. 2013;935:185-99. doi: 10.1007/978-1-62703-080-9_12.


Mammalian P4-ATPases and ABC transporters and their role in phospholipid transport.

Coleman JA, Quazi F, Molday RS.

Biochim Biophys Acta. 2013 Mar;1831(3):555-74. doi: 10.1016/j.bbalip.2012.10.006. Epub 2012 Oct 26. Review.


ABCA4 is an N-retinylidene-phosphatidylethanolamine and phosphatidylethanolamine importer.

Quazi F, Lenevich S, Molday RS.

Nat Commun. 2012 Jun 26;3:925. doi: 10.1038/ncomms1927.


Critical role of a transmembrane lysine in aminophospholipid transport by mammalian photoreceptor P4-ATPase ATP8A2.

Coleman JA, Vestergaard AL, Molday RS, Vilsen B, Andersen JP.

Proc Natl Acad Sci U S A. 2012 Jan 31;109(5):1449-54. doi: 10.1073/pnas.1108862109. Epub 2012 Jan 17.


X-linked juvenile retinoschisis: clinical diagnosis, genetic analysis, and molecular mechanisms.

Molday RS, Kellner U, Weber BH.

Prog Retin Eye Res. 2012 May;31(3):195-212. doi: 10.1016/j.preteyeres.2011.12.002. Epub 2012 Jan 3. Review.


Gene therapy: light is finally in the tunnel.

Cao H, Molday RS, Hu J.

Protein Cell. 2011 Dec;2(12):973-89. doi: 10.1007/s13238-011-1126-y. Epub 2012 Jan 10. Review.


Essential role of ELOVL4 protein in very long chain fatty acid synthesis and retinal function.

Harkewicz R, Du H, Tong Z, Alkuraya H, Bedell M, Sun W, Wang X, Hsu YH, Esteve-Rudd J, Hughes G, Su Z, Zhang M, Lopes VS, Molday RS, Williams DS, Dennis EA, Zhang K.

J Biol Chem. 2012 Mar 30;287(14):11469-80. doi: 10.1074/jbc.M111.256073. Epub 2011 Dec 24.


Lipid transport by mammalian ABC proteins.

Quazi F, Molday RS.

Essays Biochem. 2011 Sep 7;50(1):265-90. doi: 10.1042/bse0500265. Review.


Retinal degeneration 3 (RD3) protein inhibits catalytic activity of retinal membrane guanylyl cyclase (RetGC) and its stimulation by activating proteins.

Peshenko IV, Olshevskaya EV, Azadi S, Molday LL, Molday RS, Dizhoor AM.

Biochemistry. 2011 Nov 8;50(44):9511-9. doi: 10.1021/bi201342b. Epub 2011 Oct 11.


Posttranslational modifications of the photoreceptor-specific ABC transporter ABCA4.

Tsybovsky Y, Wang B, Quazi F, Molday RS, Palczewski K.

Biochemistry. 2011 Aug 16;50(32):6855-66. doi: 10.1021/bi200774w. Epub 2011 Jul 8.


Subretinal gene delivery using helper-dependent adenoviral vectors.

Wu L, Lam S, Cao H, Guan R, Duan R, van der Kooy D, Bremner R, Molday RS, Hu J.

Cell Biosci. 2011 Apr 4;1(1):15. doi: 10.1186/2045-3701-1-15.

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