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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.


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.


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.


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.


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.


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.


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.


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.


Localization of a guanylyl cyclase to chemosensory cilia requires the novel ciliary MYND domain protein DAF-25.

Jensen VL, Bialas NJ, Bishop-Hurley SL, Molday LL, Kida K, Nguyen PA, Blacque OE, Molday RS, Leroux MR, Riddle DL.

PLoS Genet. 2010 Nov 24;6(11):e1001199. doi: 10.1371/journal.pgen.1001199.


RD3, the protein associated with Leber congenital amaurosis type 12, is required for guanylate cyclase trafficking in photoreceptor cells.

Azadi S, Molday LL, Molday RS.

Proc Natl Acad Sci U S A. 2010 Dec 7;107(49):21158-63. doi: 10.1073/pnas.1010460107. Epub 2010 Nov 15.


Interaction and localization of the retinitis pigmentosa protein RP2 and NSF in retinal photoreceptor cells.

Holopainen JM, Cheng CL, Molday LL, Johal G, Coleman J, Dyka F, Hii T, Ahn J, Molday RS.

Biochemistry. 2010 Sep 7;49(35):7439-47. doi: 10.1021/bi1005249.


Oviductal glycoprotein (OVGP1, MUC9): a differentiation-based mucin present in serum of women with ovarian cancer.

Maines-Bandiera S, Woo MM, Borugian M, Molday LL, Hii T, Gilks B, Leung PC, Molday RS, Auersperg N.

Int J Gynecol Cancer. 2010 Jan;20(1):16-22. doi: 10.1111/IGC.0b013e3181bcc96d.


Knockout of GARPs and the β-subunit of the rod cGMP-gated channel disrupts disk morphogenesis and rod outer segment structural integrity.

Zhang Y, Molday LL, Molday RS, Sarfare SS, Woodruff ML, Fain GL, Kraft TW, Pittler SJ.

J Cell Sci. 2009 Apr 15;122(Pt 8):1192-200. doi: 10.1242/jcs.042531. Erratum in: J Cell Sci. 2009 Jun 1;122(Pt 11):1927.


In vivo imaging of the mouse model of X-linked juvenile retinoschisis with fourier domain optical coherence tomography.

Xu J, Molday LL, Molday RS, Sarunic MV.

Invest Ophthalmol Vis Sci. 2009 Jun;50(6):2989-93. doi: 10.1167/iovs.08-2542. Epub 2009 Jan 31.


Role of the C terminus of the photoreceptor ABCA4 transporter in protein folding, function, and retinal degenerative diseases.

Zhong M, Molday LL, Molday RS.

J Biol Chem. 2009 Feb 6;284(6):3640-9. doi: 10.1074/jbc.M806580200. Epub 2008 Dec 4.


Characterization and purification of the discoidin domain-containing protein retinoschisin and its interaction with galactose.

Dyka FM, Wu WW, Pfeifer TA, Molday LL, Grigliatti TA, Molday RS.

Biochemistry. 2008 Sep 2;47(35):9098-106. doi: 10.1021/bi800938g. Epub 2008 Aug 9.


Effect of late-stage therapy on disease progression in AAV-mediated rescue of photoreceptor cells in the retinoschisin-deficient mouse.

Janssen A, Min SH, Molday LL, Tanimoto N, Seeliger MW, Hauswirth WW, Molday RS, Weber BH.

Mol Ther. 2008 Jun;16(6):1010-7. doi: 10.1038/mt.2008.57. Epub 2008 Mar 25.


Proteomics of photoreceptor outer segments identifies a subset of SNARE and Rab proteins implicated in membrane vesicle trafficking and fusion.

Kwok MC, Holopainen JM, Molday LL, Foster LJ, Molday RS.

Mol Cell Proteomics. 2008 Jun;7(6):1053-66. doi: 10.1074/mcp.M700571-MCP200. Epub 2008 Jan 31.


Disease mechanisms and gene therapy in a mouse model for X-linked retinoschisis.

Molday LL, Min SH, Seeliger MW, Wu WW, Dinculescu A, Timmers AM, Janssen A, Tonagel F, Hudl K, Weber BH, Hauswirth WW, Molday RS.

Adv Exp Med Biol. 2006;572:283-9. No abstract available.


Prolonged recovery of retinal structure/function after gene therapy in an Rs1h-deficient mouse model of x-linked juvenile retinoschisis.

Min SH, Molday LL, Seeliger MW, Dinculescu A, Timmers AM, Janssen A, Tonagel F, Tanimoto N, Weber BH, Molday RS, Hauswirth WW.

Mol Ther. 2005 Oct;12(4):644-51.


Membrane-associated guanylate kinase proteins MPP4 and MPP5 associate with Veli3 at distinct intercellular junctions of the neurosensory retina.

Stöhr H, Molday LL, Molday RS, Weber BH, Biedermann B, Reichenbach A, Krämer F.

J Comp Neurol. 2005 Jan 3;481(1):31-41.


Role of subunit assembly in autosomal dominant retinitis pigmentosa linked to mutations in peripherin 2.

Molday RS, Molday LL, Loewen CJ.

Novartis Found Symp. 2004;255:95-112; discussion 113-6, 177-8. Review.


The heteromeric cyclic nucleotide-gated channel adopts a 3A:1B stoichiometry.

Zhong H, Molday LL, Molday RS, Yau KW.

Nature. 2002 Nov 14;420(6912):193-8.


Inactivation of the murine X-linked juvenile retinoschisis gene, Rs1h, suggests a role of retinoschisin in retinal cell layer organization and synaptic structure.

Weber BH, Schrewe H, Molday LL, Gehrig A, White KL, Seeliger MW, Jaissle GB, Friedburg C, Tamm E, Molday RS.

Proc Natl Acad Sci U S A. 2002 Apr 30;99(9):6222-7.


Expression of X-linked retinoschisis protein RS1 in photoreceptor and bipolar cells.

Molday LL, Hicks D, Sauer CG, Weber BH, Molday RS.

Invest Ophthalmol Vis Sci. 2001 Mar;42(3):816-25.


ABCR expression in foveal cone photoreceptors and its role in stargardt macular dystrophy

Molday LL, Rabin AR, Molday RS.

Am J Ophthalmol. 2000 Nov;130(5):689. No abstract available.


ABCR expression in foveal cone photoreceptors and its role in Stargardt macular dystrophy.

Molday LL, Rabin AR, Molday RS.

Nat Genet. 2000 Jul;25(3):257-8.


Rom-1 is required for rod photoreceptor viability and the regulation of disk morphogenesis.

Clarke G, Goldberg AF, Vidgen D, Collins L, Ploder L, Schwarz L, Molday LL, Rossant J, Szél A, Molday RS, Birch DG, McInnes RR.

Nat Genet. 2000 May;25(1):67-73.


Purification, characterization, and reconstitution of cyclic nucleotide-gated channels.

Molday RS, Molday LL.

Methods Enzymol. 1999;294:246-60. No abstract available.


Molecular properties of the cGMP-gated channel of rod photoreceptors.

Molday RS, Molday LL.

Vision Res. 1998 May;38(10):1315-23.


The 220-kDa rim protein of retinal rod outer segments is a member of the ABC transporter superfamily.

Illing M, Molday LL, Molday RS.

J Biol Chem. 1997 Apr 11;272(15):10303-10.


The cGMP-gated channel of the rod photoreceptor cell characterization and orientation of the amino terminus.

Molday RS, Molday LL, Dosé A, Clark-Lewis I, Illing M, Cook NJ, Eismann E, Kaupp UB.

J Biol Chem. 1991 Nov 15;266(32):21917-22.


The cGMP-gated channel of bovine rod photoreceptors is localized exclusively in the plasma membrane.

Cook NJ, Molday LL, Reid D, Kaupp UB, Molday RS.

J Biol Chem. 1989 Apr 25;264(12):6996-9.


Glycoproteins specific for the retinal rod outer segment plasma membrane.

Molday LL, Molday RS.

Biochim Biophys Acta. 1987 Feb 26;897(2):335-40.


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