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  3. Ophthalmology
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Ophthalmology

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All Publications

(140+ journal articles plus book chapters, etc.)

Links to other publications
Selected Publications, 1980 - Present
Publications Most Related to Current Research

Hazim RA, Paniagua AE, Tang L, Yang K, Kim KKO, Stiles L, Divakaruni AS, Williams DS: Vitamin B3, nicotinamide, enhances mitochondrial metabolism to promote differentiation of the retinal pigment epithelium. Journal of Biological Chemistry 298:102286, 2022.
https://www.ncbi.nlm.nih.gov/pubmed/35868562Link is external(Link is external)     PMID: 35868562.  doi: 10.1016/j.jbc.2022.102286

Chadha A, Paniagua AE, Williams DS:  Comparison of ciliary targeting of two rhodopsin-like GPCRs: role of C-terminal localization sequences in relation to cilium type. Journal of Neuroscience, in press, 2021.
PMID:   PMCID:   DOI: 10.1523/JNEUROSCI.0357-21.2021Link is external(Link is external)

Paniagua AE, Segurado A, Dolon JF, Esteve-Rudd J, Velasco A, Williams DS, Lillo C:  Key role for CRB2 in the maintenance of apicobasal polarity in retinal pigment epithelial cells. Frontiers in Cell and Developmental Biology, 9:701853, 2021.
PMID: 34262913Link is external(Link is external)  PMCID: PMC8273544Link is external(Link is external)  DOI: 10.3389/fcell.2021.701853Link is external(Link is external)

Tang C, Han J, Dalvi S, Manian K, Winschel L, Volland S, Soto CA, Galloway CA, Spencer W, Roll M, Milliner C, Bonilha VL, Johnson TB, Latchney L, Weimer JM, Augustine EF, Mink JW, Gullapalli VK, Chung M, Williams DS, and Singh R: A human model of Batten disease shows role of CLN3 in phagocytosis at the photoreceptor-RPE interface. Communications Biology, 4:161, 2021.
PMID: 33547385Link is external(Link is external)  PMCID: PMC7864947Link is external(Link is external)  DOI: 10.1038/s42003-021-01682-5Link is external(Link is external)

Hultgren NW, Fang JS, Ziegler ME, Ramirez RN, Phan DTT, Hatch MMS, Welch-Reardon KM, Paniagua AE, Kim LS, Shon NN, Williams DS, Mortazavi A, and Hughes CCW: Slug regulates the Dll4-Notch-VEGFR2 axis to control endothelial cell activation and angiogenesis. Nature Communications, 11:5400, 2020.
PMID: 33106502Link is external(Link is external)  PMCID: PMC7588439Link is external(Link is external)  DOI: 10.1038/s41467-020-18633-zLink is external(Link is external)

Segawa M, Wolf DM, Hultgren NW, Williams DS, van der Bliek AM, Shackelford DB, Liesa M, Shirihai OS: Quantification of cristae architecture reveals time-dependent characteristics of individual mitochondria. Life Science Alliance, 3(7):e201900620, 2020.
PMID: 32499316Link is external(Link is external)  PMCID: PMC7283135Link is external(Link is external)  DOI: 10.26508/lsa.201900620Link is external(Link is external)

Jiang M, Paniagua AE, Volland S, Wang H, Balaji A, Li DG, Lopes VS, Burgess BL, Williams DS: Microtubule motor transport in the delivery of melanosomes to the actin-rich apical domain of the retinal pigment epithelium. Journal of Cell Science, 133 (15): jcs242214, 2020.
PMID: 32661088Link is external(Link is external)  PMCID: PMC7420818Link is external(Link is external)  DOI: 10.1242/jcs.242214Link is external(Link is external)        

Lakkaraju A, Umapathy A, Tan LX, Daniele L, Philp NJ, Boesze-Battaglia K, Williams DS: The cell biology of the retinal pigment epithelium.
Progress in Retina and Eye Research, 78:100846, 2020.
PMID: 32105772Link is external(Link is external)  PMCID:              DOI: 10.1016/j.preteyeres.2020.100846Link is external(Link is external)

Milstein ML, Cavanaugh B, Roussey NM, Volland S, Williams DS, Goldberg AFX: Multistep peripherin-2/rds self-assembly drives membrane curvature for outer segment disk architecture and photoreceptor viability. Proceedings of the National Academy of Sciences, 117:4400-4410, 2020.
PMID: 32041874Link is external(Link is external)  PMCID: PMC7049111Link is external(Link is external)  DOI: 10.1073/pnas.1912513117Link is external(Link is external)  

Daniele LL, Caughey J, Volland S, Sharp RC, Dhingra A, Williams DS, Philp NJ, and Boesze-Battaglia K: Peroxisome turnover and diurnal modulation of antioxidant activity in retinal pigment epithelia utilizes microtubule-associated protein 1 light chain 3B. American Journal of Physiology-Cell Physiology, 317:C1194-C1204, 2019.
PMID: 31577510Link is external(Link is external)  PMCID: PMC6962520Link is external(Link is external)  DOI: 10.1152/ajpcell.00185.2019Link is external(Link is external)

Chadha A, Volland S, Baliaouri NV, Tran EM, Williams DS: The route of the visual receptor rhodopsin along the cilium.
Journal of Cell Science, 132(10):jcs229526, 2019.
PMID: 30975916Link is external(Link is external)  PMCID: PMC6550008Link is external(Link is external)  DOI: 10.1242/jcs.229526Link is external(Link is external)

Hazim RA, Volland S, Yen A, Burgess BL, Williams DS: Rapid differentiation of the human RPE cell line, ARPE-19, induced by nicotinamide.  
Experimental Eye Research, 179:18-24, 2019.
PMID: 30336127Link is external(Link is external)  PMCID: PMC6360117Link is external(Link is external)  DOI: 10.1016/j.exer.2018.10.009Link is external(Link is external)

Volland S, Williams DS: Preservation of Photoreceptor Nanostructure for Electron Tomography Using Transcardiac Perfusion Followed by High-Pressure Freezing and Freeze-Substitution. In:
Ash J, Anderson R, LaVail M, Bowes Rickman C, Hollyfield J, Grimm C. (eds)
Retinal Degenerative Diseases.  Advances in Experimental Medicine and Biology, vol. 1074. Springer, Cham, 2018.
PMID:   PMCID:   DOI: 10.1007/978-3-319-75402-4_73Link is external(Link is external)

Esteve-Rudd J, Hazim RA, Diemer T, Paniagua AE, Volland S, Umapathy A, Williams DS:
Defective phagosome motility and degradation in cell non-autonomous RPE pathogenesis of a dominant macular degeneration. Proceedings of the National Academy of Sciences, 115:5468-5473, 2018.
PMID: 29735674Link is external(Link is external)  PMCID: PMC6003516Link is external(Link is external)  DOI: 10.1073/pnas.1709211115Link is external(Link is external)

Hazim RA, Karumbayaram S, Jiang M, Dimashkie A, Lopes VS, Li D, Burgess BL, Vijayaraj P, Alva-Ornelas JA, Zack JA, Kohn DB, Gomperts BN, Pyle AD, Lowry WE, Williams DS: Differentiation of RPE cells from integration-free iPS cells and their cell biological characterization. Stem Cell Research & Therapy, 8:217, 2017.
PMID: 28969679Link is external(Link is external)  PMCID: PMC5625837Link is external(Link is external)  DOI: 10.1186/s13287-017-0652-9Link is external(Link is external)

Galloway CA, Dalvi S, Hung SSC, MacDonald LA, Latchney LR, Wong RCB, Guymer RH, Mackey DA, Williams DS, Chung MM, Gamm DM, Pebay A, Hewitt AW, Singh R: Drusen in patient-derived hiPSC-RPE models of macular dystrophies. Proceedings of the National Academy of Sciences, 114:E8214-E8223, 2017.
PMID: 28878022Link is external(Link is external)  PMCID: PMC5625924Link is external(Link is external)  DOI: 10.1073/pnas.1710430114Link is external(Link is external)

Orme MH, Liccardi G, Moderau N, Feltham R, Wicky-John S, Tenev T, Aram L, Wilson R, Bianchi K, Morris O, Monteiro Domingues C, Robertson D, Tare M, Wepf A, Williams D, Bergmann A, Gstaiger M, Arama E, Ribeiro PS, Meier P: The unconventional myosin CRINKLED and its mammalian orthologue MYO7A regulate caspases in their signaling roles. Nature Communications, 7:10972 (1-12), 2016.
PMID: 26960254Link is external(Link is external)  PMCID: PMC4792956Link is external(Link is external)  DOI: 10.1038/ncomms10972Link is external(Link is external)

Goldberg AG, Moritz OL, Williams DS:  Molecular basis for photoreceptor outer segment architecture. Progress in Retinal and Eye Research, 55:52-81, 2016.
PMID: 27260426Link is external(Link is external)  PMCID: PMC5112118Link is external(Link is external)  DOI: 10.1016/j.preteyeres.2016.05.003Link is external(Link is external)

Williams DS, Chadha A, Hazim R, Gibbs D:  Gene Therapy approaches for prevention of retinal degeneration in Usher syndrome. Gene Therapy, 24:68-71, 2018.
PMID: 28054582Link is external(Link is external)  PMCID: PMC6055994Link is external(Link is external)  DOI: 10.1038/gt.2016.81Link is external(Link is external)

Volland S, Hughes LC, Kong C, Burgess BL, Linberg KA, Luna G, Zhou ZH, Fisher SK, and Williams DS: Three-dimensional organization of nascent rod outer segment disk membranes. Proceedings of the National Academy of Sciences, 112(48):14870-14875, 2015.
PMID: 26578801Link is external(Link is external)  PMCID: PMC4672767Link is external(Link is external)  DOI: 10.1073/pnas.1516309112Link is external(Link is external)

Jiang M, Esteve-Rudd J, Lopes VS, Diemer T, Lillo C, Rump A, Williams DS: Microtubule motors transport phagosomes in the RPE and lack of KLC1 leads to AMD-like pathogenesis. Journal of Cell Biology, 210(4):595-611, 2015.
PMID: 26261180Link is external(Link is external)  PMCID: PMC4539993Link is external(Link is external)  DOI: 10.1083/jcb.201410112Link is external(Link is external)

Volland S, Esteve-Rudd J, Hoo J, Yee C, Williams DS: A comparison of some organizational characteristics of the mouse central retina and the human macula. PLos One, 10:e0125631, 2015.
PMID: 25923208Link is external(Link is external)  PMCID: PMC4414478Link is external(Link is external)  DOI: 10.1371/journal.pone.0125631Link is external(Link is external)

Eblimit A, Nguyen TM, Chen Y, Esteve-Rudd J, Zhong H, Letteboer S, Van Reeuwijk J, Simons DL, Ding Q, Wu KM, Li Y, Van Beersum S, Moayedi Y, Xu H, Pickard P, Wang K, Gan L, Wu SM, Williams DS, Mardon G, Roepman R, Chen R: Spata7 is a retinal ciliopathy gene critical for correct RPGRIP1 localization and protein trafficking in the retina. Human Molecular Genetics, 24:1584-1601, 2015.
PMID: 25398945Link is external(Link is external)  PMCID: PMC4351378Link is external(Link is external)  DOI: 0.1093/hmg/ddu573Link is external(Link is external)

Frost LS, Lopes VS, Bragin A, Reyes-Reveles J, Brancato J, Cohen A, Mitchell CH, Williams DS, Boesze-Battaglia K: The contribution of melanoregulin to microtubule-associated protein 1 light chain 3 (LC3) associated phagocytosis in retinal pigment epithelium. Molecular Neurobiology, 52(3):1135-1151, 2014.
PMID: 25301234Link is external(Link is external)  PMCID: PMC5531606Link is external(Link is external)  DOI: 10.1007/s12035-014-8920-5Link is external(Link is external)

Gee HY, Ashraf S, Wan X, Vega-Warner V, Esteve-Rudd J, Lovric S, Fang H, Hurd TW, Sadowski CE, Allen SJ, Otto EA, Korkmaz E, Washburn J, Levy S, Williams DS, Bakkaloglu SA, Zolotnitskaya A, Ozaltin F, Zhou W, Hildebrandt F: Mutations in EMP2 cause childhood-onset nephrotic syndrome.  American Journal of Human Genetics, 94:884-90, 2014.
PMID: 24814193Link is external(Link is external)  PMCID: PMC4121470Link is external(Link is external)  DOI: 10.1016/j.ajhg.2014.04.010Link is external(Link is external)

Crouse JA, Lopes VS, SanAgustin JT, Keady BT, Williams DS, Pazour GJ: Distinct functions for IFT140 and IFT20 in opsin transport. Cytoskeleton, 71:302-310, 2014.
PMID: 24619649Link is external(Link is external)  PMCID: PMC4173073Link is external(Link is external)  DOI: 10.1002/cm.21173Link is external(Link is external)

Almenar-Queralt A, Falzone TL, Shen Z, Lillo C, Killian RL, Arreola AS, Niederst ED, Ng KS, Kim SN, Briggs SP, Williams DS, Goldstein LS: UV irradiation accelerates amyloid precursor protein (APP) processing and disrupts APP axonal transport. Journal of Neuroscience, 34:3320-39, 2014.
PMID: 24573290Link is external(Link is external)  PMCID: PMC3935090Link is external(Link is external)  DOI: 10.1523/JNEUROSCI.1503-13.2014Link is external(Link is external)

Mei H, Gonzalez S, Nakatsu MN, Baclagon ER, Lopes VS, Williams DS, Deng SX: A Three-dimensional culture method to expand limbal stem/progenitor cells. Tissue engineering Part C, Methods, 2014.
PMID: 24047104Link is external(Link is external)  PMCID: PMC4005490Link is external(Link is external)  DOI: 10.1089/ten.tec.2013.0246Link is external(Link is external)

Ashraf S, Gee HY, Woerner S, Xie LX, Vega-Warner V, Lovric S, Fang H, Song X, Cattran DC, Avila-Casado C, Paterson AD, Nitschké P, Bole-Feysot C, Cochat P, Esteve-Rudd J, Haberberger B, Allen SJ, Zhou W, Airik R, Otto EA, Barua M, Al-Hamed MH, Kari JA, Evans J, Bierzynska A, Saleem MA, Böckenhauer D, Kleta R, El Desoky S, Hacihamdioglu DO, Gok F, Washburn J, Wiggins RC, Choi M, Lifton RP, Levy S, Han Z, Salviati L, Prokisch H, Williams DS, Pollak M, Clarke CF, Pei Y, Antignac C, Hildebrandt F: ADCK4 mutations promote steroid-resistant nephrotic syndrome through CoQ10 biosynthesis disruption. Journal of Clinical Investigation, 123(12):5179-89, 2013.
PMID: 24270420Link is external(Link is external)  PMCID: PMC3859425Link is external(Link is external)  DOI: 10.1172/JCI69000Link is external(Link is external)

Zariwala MA, Gee HY, Kurkowiak M, Al-Mutairi DA, Leigh MW, Hurd TW, Hjeij R, Dell SD, Chaki M, Dougherty GW, Adan M, Spear PC, Esteve-Rudd J, Loges NT, Rosenfeld M, Diaz KA, Olbrich H, Wolf WE, Sheridan E, Batten TF, Halbritter J, Porath JD, Kohl S, Lovric S, Hwang DY, Pittman JE, Burns KA, Ferkol TW, Sagel SD, Olivier KN, Morgan LC, Werner C, Raidt J, Pennekamp P, Sun Z, Zhou W, Airik R, Natarajan S, Allen SJ, Amirav I, Wieczorek D, Landwehr K, Nielsen K, Schwerk N, Sertic J, Kohler G, Washburn J, Levy S, Fan S, Koerner-Rettberg C, Amselem S, Williams DS, Mitchell BJ, Drummond IA, Otto EA, Omran H, Knowles MR, Hildebrandt F: ZMYND10 Is Mutated in Primary Ciliary Dyskinesia and Interacts with LRRC6.
American Journal of Human Genetics, 93:336-345, 2013.
PMID: 23891469Link is external(Link is external)  PMCID: PMC3738827Link is external(Link is external)  DOI: 10.1016/j.ajhg.2013.06.007Link is external(Link is external)

Frost LS, Lopes VS, Stefano FP, Bragin A, Williams DS, Mitchell CH, Boesze-Battaglia K: Loss of Melanoregulin (MREG) enhances Cathepsin-D secretion by the Retinal Pigment Epithelium (RPE). Visual Neuroscience, 30:55-64. 2013.
PMID: 23611523Link is external(Link is external)  PMCID: PMC3912860Link is external(Link is external)  DOI: 10.1017/S0952523813000096Link is external(Link is external)

Lopes VS, Boye SE, Louie CM, Boye S, Dyka F, Chiodo V, Fofo H, Hauswirth WW, Williams DS: Retinal gene therapy with a large MYO7A cDNA using Adeno-associated virus. Gene Therapy, 20:824-833 2013.
PMID: 23344065Link is external(Link is external)  PMCID: PMC3640772Link is external(Link is external)  DOI: 10.1038/gt.2013.3Link is external(Link is external)

Strong R, Miller RA, Astle CM, Baur JA, de Cabo R, Fernandez E, Guo W, Javors M, Kirkland JL, Nelson JF, Sinclair DA, Teter B, Williams D, Zaveri N, Nadon NL, Harrison DE: Evaluation of resveratrol, green tea extract, curcumin, oxaloacetic acid, and medium-chain triglyceride oil on life span of genetically heterogeneous mice. Journal of Gerontology A Biological Sciences and Medical Sciences, 68:6-16, 2013.
PMID: 22451473Link is external(Link is external)  PMCID: PMC3598361Link is external(Link is external)  DOI: 10.1093/gerona/gls070Link is external(Link is external)

Trivedi D, Colin E, Louie CM, Williams DS: Live-cell imaging evidence for the ciliary transport of rod photoreceptor opsin by heterotrimeric kinesin-2. Journal of Neuroscience, 32:10587-10593, 2012.
PMID: 22855808Link is external(Link is external)  PMCID: PMC3428073Link is external(Link is external)  DOI: 10.1523/JNEUROSCI.0015-12.2012Link is external(Link is external)

Da Cruz S, Parone PA, Lopes VS, Lillo C, McAlonis-Downes M, Lee SK, Vetto AP, Petrosyan S, Marsala MS, Murphy AN, Williams DS, Spiegelman BM, Cleveland DW: Elevated PGC-1α activity sustains mitochondrial biogenesis and muscle function without extending survival in a mouse model of inherited ALS. Cell Metabolism, 15:778-786, 2012.
PMID: 22560226Link is external(Link is external)  PMCID: PMC3565468Link is external(Link is external)  DOI: 10.1016/j.cmet.2012.03.019Link is external(Link is external)

Engelhardt M, Tosha C, Lopes VS, Chen B, Nguyen L, Nusinowitz S, Williams DS: Functional and morphological analysis of the subretinal injection of RPE cells. 
Visual Neuroscience, 29:83-93 (plus front cover), 2012.
PMID: 22391151Link is external(Link is external)  PMCID: PMC3694443Link is external(Link is external)  DOI: 10.1017/S0952523812000041Link is external(Link is external)

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: Essential role of ELOVL4 in very long chain fatty acid synthesis and retinal function. Journal of Biological Chemistry, 287:11469-11480, 2012.
PMID: 22199362Link is external(Link is external)  PMCID: PMC3322886Link is external(Link is external)  DOI: 10.1074/jbc.M111.256073Link is external(Link is external)

Williams DS, Lopes VS: The many different cellular functions of MYO7A in the retina. Biochemical Society transactions, 39:1207-10, 2011.
PMID: 21936790Link is external(Link is external)  PMCID: PMC3703834Link is external(Link is external)  DOI: 10.1042/BST0391207Link is external(Link is external)

Jacobson SG, Cideciyan AV, Gibbs D, Sumaroka A, Roman AJ, Aleman TS, Schwartz SB, Olivares MB, Russell RC, Steinberg JD, Kenna MA, Kimberling WJ, Rehm HL, Williams DS: Retinal Disease Course in Usher Syndrome 1B due to MYO7A Mutations. Investigative Ophthalmology & Visual Science, 52:7924-7936, 2011.
PMID: 21873662Link is external(Link is external)  PMCID: PMC3263772Link is external(Link is external)  DOI: 10.1167/iovs.11-8313Link is external(Link is external)

Phillips JB, Blanco-Sanchez B, Lentz JJ, Tallafuss A, Khanobdee K, Sampath S, Jacobs ZG, Han PF, Mishra M, Williams DS, Keats BJ, Washbourne P, Westerfield M: Harmonin (Ush1c) is required in zebrafish Muller glial cells for photoreceptor synaptic development and function. Disease Models & Mechanisms, 4:786-800, 2011.
PMID: 21757509Link is external(Link is external)  PMCID: PMC3209648Link is external(Link is external)  DOI: 10.1242/dmm.006429Link is external(Link is external)

Lopes VS, Gibbs D, Libby RT, Aleman TS, Welch DL, Lillo C, Jacobson SG, Radu RA, Steel KP, Williams DS: The Usher 1B protein, MYO7A, is required for normal localization and function of the visual retinoid cycle enzyme, RPE65. Human Molecular Genetics, 20:2560-2570, 2011.
PMID: 21493626Link is external(Link is external)  PMCID: PMC3110002Link is external(Link is external)  DOI: 10.1093/hmg/ddr155Link is external(Link is external)

Lopes VS, Jimeno D, Khanobdee K, Song X, Chen B, Nusinowitz S, Williams DS: Dysfunction of heterotrimeric kinesin-2 in rod photoreceptor cells and the role of opsin mislocalization in rapid cell death. Molecular Biology of the Cell, 21:4076-4088 (plus front cover), 2010.
PMID: 20926680Link is external(Link is external)  PMCID: PMC2993738Link is external(Link is external)  DOI: 10.1091/mbc.E10-08-0715Link is external(Link is external)

Otto EA, Hurd TW, Airik R, Chaki M, Zhou W, Stoetzel C, Patil SB, Levy S, Ghosh AK, Murga-Zamalloa CA, van Reeuwijk J, Letteboer SJ, Sang L, Giles RH, Liu Q, Coene KL, Estrada-Cuzcano A, Collin RW, McLaughlin HM, Held S, Kasanuki JM, Ramaswami G, Conte J, Lopez I, Washburn J, Macdonald J, Hu J, Yamashita Y, Maher ER, Guay-Woodford LM, Neumann HP, Obermuller N, Koenekoop RK, Bergmann C, Bei X, Lewis RA, Katsanis N, Lopes V, Williams DS, Lyons RH, Dang CV, Brito DA, Dias MB, Zhang X, Cavalcoli JD, Nurnberg G, Nurnberg P, Pierce EA, Jackson PK, Antignac C, Saunier S, Roepman R, Dollfus H, Khanna H, Hildebrandt F: Candidate exome capture identifies mutation of SDCCAG8 as the cause of a retinal-renal ciliopathy. Nature Genetics, 42:840-850, 2010.
PMID: 20835237Link is external(Link is external)  PMCID: PMC2947620Link is external(Link is external)  DOI: 10.1038/ng.662Link is external(Link is external)

Liao JL,Yu J, Huang K, Hu J, Diemer T, Ma Z, Dvash T, Yang XJ, Travis GH, Williams DS, Bok D, Fan G: Molecular signature of primary retinal pigment epithelium and stem-cell-derived RPE cells. Human Molecular Genetics, 19:4229-4238, 2010.
PMID: 20709808Link is external(Link is external)  PMCID: PMC3115666Link is external(Link is external)  DOI: 10.1093/hmg/ddq341Link is external(Link is external)

Louie CM, Caridi G, Lopes VS, Brancati F, Kispert A, Lancaster MA, Schlossman AM, Otto EA, Leitges M, Grone HJ, Lopez I, Gudiseva HV, O'Toole JF, Vallespin E, Ayyagari R, Ayuso C, Cremers FP, den Hollander AI, Koenekoop RK, Dallapiccola B, Ghiggeri GM, Hildebrandt F, Valente EM, Williams DS, Gleeson JG: AHI1 is required for photoreceptor outer segment development and is a modifier for retinal degeneration in nephronophthisis. Nature Genetics, 42:175-180, 2010.
PMID: 20081859Link is external(Link is external)  PMCID: PMC2884967Link is external(Link is external)  DOI: 10.1038/ng.519Link is external(Link is external)

Gibbs D, Diemer T, Khanobdee K, Hu J, Bok D, Williams DS: Function of MYO7A in the human RPE and the validity of shaker1 mice as a model for Usher syndrome 1B.
Investigative Ophthalmology and Visual Science, 51:1130-1135, 2010.
PMID: 19643958Link is external(Link is external)  PMCID: PMC2868451Link is external(Link is external)  DOI: 10.1167/iovs.09-4032Link is external(Link is external)

Schwander M, Lopes V, Sczaniecka A, Gibbs D, Lillo C, Delano D, Tarantino LM, Wiltshire T, Williams DS, Müller U: A novel allele of myosin VIIa reveals a critical function for the C-terminal FERM domain for melanosome transport in retinal pigment epithelial cells. Journal of Neuroscience, 29:15810-15818, 2009.
PMID: 20016096Link is external(Link is external)  PMCID: PMC2834289Link is external(Link is external)  DOI: 10.1523/JNEUROSCI.4876-09.2009Link is external(Link is external)

Abe N, Almenar-Queralt A, Lillo C, Shen Z, Lozach J, Briggs SP, Williams DS, Goldstein LS, Cavalli V: Sunday Driver interacts with two distinct classes of axonal organelles. Journal of Biological Chemistry, 284:34628-34639, 2009.
PMID: 19801628Link is external(Link is external)  PMCID: PMC2787325Link is external(Link is external)  DOI: 10.1074/jbc.M109.035022Link is external(Link is external)

Avasthi P, Watt CB, Williams DS, Le YZ, Li S, Chen C-K, Marc RE, Frederick JM, Baehr W: Trafficking of membrane proteins to cone but not rod outer segments is dependent on heterotrimeric kinesin-II. Journal of Neuroscience, 29:14287-14298, 2009.
PMID: 19906976Link is external(Link is external)  PMCID: PMC2788486Link is external(Link is external)  DOI: 10.1523/JNEUROSCI.3976-09.2009Link is external(Link is external)

Williams DS, Cash A, Hamadani L, Diemer T: Oxaloacetate supplementation increases lifespan in Caenorhabditis elegans through an AMPK/FOXO-dependent pathway. Aging Cell, 8:765-768, 2009.
PMID: 19793063Link is external(Link is external) PMCID: PMC2988682Link is external(Link is external)  DOI: 10.1111/j.1474-9726.2009.00527.xLink is external(Link is external)

Grillet N, Xong W, Reynolds A, Kazmierczak P, Sato T, Lillo C, Dumont RA, Hintermann E, Sczaniecka A, Schwander M, Williams DS, Kachar B, Gillespie PG, Mueller U: Harmonin mutations cause mechanotransduction defects in cochlear hair cells. Neuron, 62:375-387, 2009.
PMID: 19447093Link is external(Link is external)  PMCID: PMC2691393Link is external(Link is external)  DOI: 10.1016/j.neuron.2009.04.006Link is external(Link is external)

Falzone TL, Stokin GB, Lillo C, Rodrigues EM, Westerman EL, Williams DS, Goldstein LSB: Axonal stress kinase activation and tau misbehavior induced by kinesin-1 transport defects. Journal of Neuroscience, 29:5758-5767, 2009.
PMID: 19420244Link is external(Link is external)  PMCID: PMC3849468Link is external(Link is external)  DOI: 10.1523/JNEUROSCI.0780-09.2009Link is external(Link is external)

Gibbs D, Cideciyan AV, Jacobson SG, Williams DS: Retinal pigment epithelium defects in humans and mice with mutations in MYO7A: imaging melanosome-specific autofluorescence. Investigative Ophthalmology and Visual Science, 50:4386-4393, 2009.
PMID: 19324852Link is external(Link is external)  PMCID: PMC2884175Link is external(Link is external)  DOI: 10.1167/iovs.09-3471Link is external(Link is external)

Williams DS, Aleman TS, Lillo C, Lopes VS, Hughes LC, Stone EM, Jacobson SG: Harmonin in the murine retina and the retinal phenotypes of Ush1c-mutant mice and human USH1C. Investigative Ophthalmology and Visual Science, 50:3881-3889, 2009.
PMID: 19324851Link is external(Link is external) PMCID: PMC2893298Link is external(Link is external)  DOI: 10.1167/iovs.08-3358Link is external(Link is external)

Damek-Poprawa M, Diemer T, Lopes VS, Lillo C, Harper, DC, Marks MS, Wu Y, Sparrow JR, Rachel R, Williams DS, Boesze-Battaglia K: Melanoregulin (MREG) modulates lysosome function in pigment epithelial cells. Journal of Biological Chemistry, 284:10877-10889, 2009.
PMID: 19240024Link is external(Link is external) PMCID: PMC2667774Link is external(Link is external)  DOI: 10.1074/jbc.M808857200Link is external(Link is external)

Stokin GB, Almenar-Queralt A, Gunawardena S, Rodrigues EM, Falzone T, Kim J, Lillo C, Mount SL, Roberts EA, McGowan E, Williams DS, Goldstein LSB: Amyloid precursor protein-induced axonopathies are independent of amyloid-beta peptides. Human Molecular Genetics, 17:3474-3486, 2008.
PMID: 18694898Link is external(Link is external)  PMCID: PMC2722897Link is external(Link is external)  DOI: 10.1093/hmg/ddn240Link is external(Link is external)

Yang Z*, Chen Y, Lillo C, Chien J, Yu Z, Michaelides M, Klein M, Howes KA, Li Y, Kaminoh Y, Chen H, Zhao C, Al-Sheikh YT, Karan G, Corbeil D, Escher P, Kamaya S, Li C, Johnson S, Frederick JM, Zhao Y, Wang C, Cameron DJ, Huttner WB, Schorderet DF, Munier FL, Moore AT, Birch DG, Baehr W, Hunt DM, Williams DS*, Zhang, K*: Mutant prominin 1 found in patients with macular degeneration disrupts photoreceptor disk morphogenesis in mice. Journal of Clinical Investigation, 118:2908-2916, 2008. *Co-corresponding authors.
PMID: 18654668Link is external(Link is external)  PMCID: PMC2483685Link is external(Link is external)  DOI: 10.1172/JCI35891Link is external(Link is external)

Jacobson SG, Cideciyan AV, Aleman TS, Sumaroka A, Roman AJ, Gardner LM, Prosser HM, Mishra M, Bech-Hansen T, Herrera W, Schwartz SB, Liu X-Z, Kimberling WJ, Steel KP, Williams DS: Usher syndromes due to MYO7A, PCDH15, USH2A or GPR98 mutations share retinal disease mechanism. Human Molecular Genetics, 17:2405-2415, 2008.
PMID: 18463160Link is external(Link is external)  PMCID: PMC2733815Link is external(Link is external)  DOI: 10.1093/hmg/ddn140Link is external(Link is external)

Allocca M, Doria M, Petrillo M, Colella P, Garcia-Hoyos M, Gibbs D, Kim SR, Maguire A, Rex TS, Vicino U, Cutillo L, Sparrow JR, Williams DS, Bennett J, Auricchio A: Serotype-dependent packaging of large genes in adeno-associated viral vectors results in effective gene delivery in mice. Journal of Clinical Investigation, 118:1955-1964, 2008.
PMID: 18414684Link is external(Link is external)  PMCID: PMC2298836Link is external(Link is external)  DOI: 10.1172/JCI34316Link is external(Link is external)

Feathers KL, Lyubarsky AL, Khan NW, Teofilo K, Swaroop A, Williams DS, Pugh EN, Thompson DA: Nrl-knockout mice deficient in Rpe65 fail to synthesize 11-cis retinal and cone outer segments. Investigative Ophthalmology and Visual Science, 49:1126-1135, 2008.
PMID: 18326740Link is external(Link is external)  PMCID: PMC4505835Link is external(Link is external)  DOI: 10.1167/iovs.07-1234Link is external(Link is external)

Williams DS: Usher syndrome: animal models, retinal function of Usher proteins, and prospects for gene therapy. Vision Research, 48:433-441, 2008.
PMID: 17936325Link is external(Link is external)  PMCID: PMC2680226Link is external(Link is external)  DOI: 10.1016/j.visres.2007.08.015Link is external(Link is external)

Klomp AE, Teofilo K, Legacki E, Williams DS: Analysis of the linkage of MYRIP and MYO7A to melanosomes by RAB27A in retinal pigment epithelial cells. Cell Motility and the Cytoskeleton, 64:474-487 (plus front cover), 2007.
PMID: 17352418Link is external(Link is external)  PMCID:   DOI: 10.1002/cm.20198Link is external(Link is external)

Hashimoto T, Gibbs D, Lillo C, Azarian SM, Legacki E, Zhang X-M, Yang XJ, Williams DS: Lentiviral gene replacement therapy of retinas in a mouse model for Usher syndrome type 1B. Gene Therapy, 14:584-594, 2007.
PMID: 17268537Link is external(Link is external)  PMCID:   DOI: 10.1038/sj.gt.3302897Link is external(Link is external)

Boesze-Battaglia K, Song H, Sokolov M, Lillo C, Pankoski-Walker L, Gretzula C, Gallagher B, Rachel RA, Jenkins NA, Copeland NG, Morris F, Yeagle P, Williams DS, Damek-Poprawa M: The tetraspanin protein, peripherin-2, complexes with melanoregulin, a putative membrane fusion regulator. Biochemistry, 46:1256-1272, 2007.
PMID: 17260955Link is external(Link is external)  PMCID: PMC4472003Link is external(Link is external)  DOI: 10.1021/bi061466iLink is external(Link is external)

Schubert D, Williams DS: ‘Cisgenic’ as a product designation. Nature Biotechnology, 24:1327-1329, 2006.
PMID: 17093469Link is external(Link is external)  PMCID:   DOI: 10.1038/nbt1106-1327Link is external(Link is external)

Jimeno D, Feiner L, Lillo C, Teofilo K, Goldstein LSB, Pierce E, Williams DS: Analysis of kinesin-2 function in photoreceptor cells using synchronous Cre-loxP knockout of Kif3a with RHO-Cre. Investigative Ophthalmology and Visual Science, 47:5039-5046, 2006.
PMID: 17065525Link is external(Link is external)  PMCID: PMC1904505Link is external(Link is external)  DOI: 10.1167/iovs.06-0032Link is external(Link is external)

Chang B, Khanna H, Hawes N, Jimeno, D, He S, Lillo, C, Parapuram SK, Cheng H, Scott A, Hurd RE, Sayer JA, Otto EA, Attanasio M, O’Toole JF, Jin G, Shou C, Hildebrandt F, Williams DS, Heckenlively JR, Swaroop A: In-frame deletion in a novel centrosomal/ciliary protein CEP290/NPHP6 perturbs its interaction with RPGR and results in early-onset retinal degeneration in the rd16 mouse. Human Molecular Genetics, 15:1847-1857, 2006.
PMID: 16632484Link is external(Link is external)  PMCID: PMC1592550Link is external(Link is external)  DOI: 10.1093/hmg/ddl107Link is external(Link is external)

Sayer JA, Otto EA, O’Toole JF, Nurnberg G, Kennedy MA, Becker C, Hennies HC, Helou J, Attanasio M, Fausett BV, Utsch B, Khanna H, Liu Y, Drummond I, Kawakami I, Kusakabe T, Tsuda M, Ma L, Lee H, Larson RG, Allen SJ, Wilkinson CJ, Nigg EA, Shou C, Lillo C, Williams DS, Hoppe B, Kemper M, Neuhaus T, Parisi MA, Glass IA, Petry M, Kispert A, Zou Y, Gloy J, Ganner A, Walz G, Zhu X, Goldman D, Nurnberg P, Swaroop A, Leroux MR, Hildebrandt F: The centrosomal protein, nephrocystin-6, is mutated in Joubert syndrome and activates transcription factor ATF4. Nature Genetics, 38:674-681, 2006.
PMID: 16682973Link is external(Link is external)  PMCID:   DOI: 10.1038/ng1786Link is external(Link is external)

Senften M, Schwander M, Kazmierczak P, Lillo C, Shin J-B, Hasson T, Géléoc GSG, Gillespie P, Williams DS, Holt J, Müller U: Physical and functional interaction between protocadherin 15 and myosin VIIa in mechanosensory hair cells. Journal of Neuroscience, 26:2060-2071, 2006.
PMID: 16481439Link is external(Link is external)  PMCID: PMC2712835Link is external(Link is external)  DOI: 10.1523/JNEUROSCI.4251-05.2006Link is external(Link is external)

Azarian SM, McLeod I, Lillo C, Gibbs D, Yates JR, Williams DS: Proteomic analysis of mature melanosomes from the retinal pigmented epithelium. Journal of Proteome Research, 5:521-529, 2006.
PMID: 16512666Link is external(Link is external)  PMCID:   DOI: 10.1021/pr0502323Link is external(Link is external)

Jimeno D, Lillo C, Roberts EA, Goldstein LSB, Williams DS: Kinesin-2 and photoreceptor cell death: requirement of motor subunits. Experimental Eye Research, 82:351-353, 2006.
PMID: 16337628Link is external(Link is external)  PMCID:   DOI: 10.1016/j.exer.2005.10.026Link is external(Link is external)

Zhu X, Wu K, Rife L, Cawley NX, Brown B, Adams T, Teofilo K, Lillo C, Williams DS, Loh YP, Craft CM: Carboxypeptidase E is required for normal synaptic transmission from photoreceptors to the inner retina. Journal of Neurochemistry, 95:1351-1362, 2005.
PMID: 16219026Link is external(Link is external)  PMCID:   DOI: 10.1111/j.1471-4159.2005.03460.xLink is external(Link is external)

Khanna H, Hurd TW, Lillo C, Shu X, Parapuram SK, He S, Akimoto M, Wright AF, Margolis B, Williams DS, Swaroop A: RPGR-ORF15, which is mutated in retinitis pigmentosa, associates with SMC1, SMC3, and microtubule transport proteins. Journal of Biological Chemistry, 280:33580-33587, 2005.
PMID: 16043481Link is external(Link is external)  PMCID: PMC1249479Link is external(Link is external)  DOI: 10.1074/jbc.M505827200Link is external(Link is external)

Kitamoto J, Libby RT, Gibbs D, Steel KP, Williams DS: Myosin VI is required for normal retinal function. Experimental Eye Research, 81:116-120, 2005.
PMID: 15978262Link is external(Link is external)  PMCID:   DOI: 10.1016/j.exer.2005.02.014Link is external(Link is external)

Karan G, Lillo C, Yang Z, Cameron J, Locke KG, Zhao Y, Thirumalaichary S, Li C, Birch DG, Vollmer-Sanar H, Williams DS*, Zhang K*: Lipofuscin accumulation, abnormal electrophysiology and photoreceptor degeneration in mutant ELOVL4 transgenic mice: a model for macular degeneration. Proceedings of the National Academy Sciences, 102:4164-4169, 2005. *Co-corresponding authors.
PMID: 15749821Link is external(Link is external)  PMCID: PMC554798Link is external(Link is external)  DOI: 10.1073/pnas.0407698102Link is external(Link is external)

Stokin GB, Lillo C, Falzone T, Brusch RG, Rockenstein E, Mount S, Raman R, Davies P, Masliah E, Williams DS, Goldstein LSB: Axonpathy and transport deficits in the pathogenesis of Alzheimer’s disease. Science, 307:1282-1288, 2005.
PMID: 15731448Link is external(Link is external)  PMCID:   DOI: 10.1126/science.1105681Link is external(Link is external)

Otto EA, Loeys B, Khanna H, Hellemans J, Sudbrak R, Fan S, Muerb U, O’Toole JF, Helou J, Attanasio M, Utsch B, Sayer JA, Lillo C, Jimeno D, Couck P, De Paepe A, Reinhardt R, Klages S, Ysuda M, Kawakami I, Kusakabe T, Omran H, ImmA, Tippens M, Raymond PA, Hill J, Beales P, He S, Kispert A, Margolis B, Williams DS, Swaroop A, Hildebrandt F: Nephrocystin-5, a ciliary IQ domain protein, is mutated in Senior-Loken syndrome and interacts with RPGR and calmodulin. Nature Genetics, 37:282-288, 2005.
PMID: 15723066Link is external(Link is external)  PMCID:   DOI: 10.1038/ng1520Link is external(Link is external)

Daniele L, Lillo C, Lyubarsky A, Philp N, Mears AJ, Swaroop A, Williams DS, Pugh E: Cone-like morphological, molecular and functional features of the photoreceptors of the Nrl-/- knockout mouse. Investigative Ophthalmology and Visual Science, 46:2156-2167, 2005.
PMID: 15914637Link is external(Link is external)  PMCID: PMC4505833Link is external(Link is external)  DOI: 10.1167/iovs.04-1427Link is external(Link is external)

Gibbs D, Azarian SA, Lillo C, Klomp AEM, Kitamoto J, Steel KP, Libby RT, Williams DS: Role of myosin VIIa and Rab27a in the motility and localization of RPE melanosomes. Journal of Cell Science, 117:6473-6483, 2004.
PMID: 15572405Link is external(Link is external)  PMCID: PMC2942070Link is external(Link is external)  DOI: 10.1242/jcs.01580Link is external(Link is external)

Libby RT, Lillo C, Kitamoto J, Williams DS, Steel KP: Myosin Va is required for normal photoreceptor synaptic activity. Journal of Cell Science, 117:4509-4515, 2004.
PMID: 15316067Link is external(Link is external)  PMCID:   DOI: 10.1242/jcs.01316Link is external(Link is external)

Liu J, Lillo C, Jonsson PA, Ward CM, Subramaniam JR, Rothstein JD, Marklund S, Andersen PM, Brännström T, Wong PC, Williams DS, Cleveland DW: Toxicity of familial ALS-linked SOD1 mutants from selective recruitment to spinal mitochondria. Neuron, 43:5-17, 2004.
PMID: 15233913Link is external(Link is external)  PMCID:   DOI: 10.1016/j.neuron.2004.06.016Link is external(Link is external)

Siemens J, Lillo C, Dumont RA, Williams DS, Gillespie PG, Müller U: Cadherin 23 is a component of the tip link in hair cell stereocilia. Nature, 428:950-955, 2004.
PMID: 15057245Link is external(Link is external)  PMCID:  DOI: 10.1038/nature02483Link is external(Link is external)

Libby RT, Kitamoto J, Holme RH, Williams DS, Steel KP: Cdh23 mutations in the mouse associated with retinal dysfunction but not retinal degeneration. Experimental Eye Research, 77:731-739, 2003.
PMID: 14609561Link is external(Link is external)  PMCID:   DOI: 10.1016/j.exer.2003.07.007Link is external(Link is external)

Adams RA, Liu X, Williams DS, Newton AC: Differential spatial and temporal phosphorylation of the visual receptor, rhodopsin, at its two primary phosphorylation sites in mice exposed to light.  Biochemical Journal, 374:537-543, 2003.
PMID: 12809555Link is external(Link is external)  PMCID: PMC1223618Link is external(Link is external)   DOI: 10.1042/BJ20030408Link is external(Link is external)

Gibbs D, Kitamoto J, Williams DS: Abnormal phagocytosis by retinal pigmented epithelium that lacks myosin VIIa, the Usher syndrome 1B protein. Proceedings of the National Academy Sciences, 100:6481-6486, 2003.
PMID: 12743369Link is external(Link is external)  PMCID: PMC164472Link is external(Link is external)  DOI: 10.1073/pnas.1130432100Link is external(Link is external)

Xia C, Roberts EA, Her L, Liu X, Williams DS, Cleveland, DW, Goldstein LS: Abnormal neurofilament transport caused by targeted disruption of neuronal kinesin heavy chain KIF5A. Journal of Cell Biology, 161:55-66, 2003.
PMID: 12682084Link is external(Link is external)  PMCID: PMC2172877Link is external(Link is external)  DOI: 10.1083/jcb.200301026Link is external(Link is external)

Udovichenko IP, Gibbs D, Williams DS: Actin-based motor properties of native myosin VIIa.  Journal of Cell Science, 115:445-450, 2002.
PMID: 11839794Link is external(Link is external)  PMCID:  DOI:

Williams DS: Transport to the photoreceptor outer segment by myosin VIIa and kinesin II. Vision Research, 42:455-462, 2002.
PMID: 11853761Link is external(Link is external)  PMCID:   DOI: 10.1016/s0042-6989(01)00228-0Link is external(Link is external)

Liu X, Williams DS: Coincident onset of expression of myosin VIIa and opsin in the cilium of the developing photoreceptor cell. Experimental Eye Research, 72:351-355, 2001.
PMID: 11180984Link is external(Link is external)  PMCID:   DOI: 10.1006/exer.2000.0963Link is external(Link is external)

Marszalek JR, Liu X, Roberts E, Chui D, Marth J, Williams DS, Goldstein LSB: Genetic evidence for a kinesin-II mediated pathway for opsin and arrestin transport in mammalian photoreceptors. Cell, 102:175-187, 2000.
PMID: 10943838Link is external(Link is external)  PMCID:   DOI: 10.1016/s0092-8674(00)00023-4Link is external(Link is external)

Liu X, Udovichenko, IP, Brown SDM, Steel KP, Williams DS: Myosin VIIa participates in opsin transport through the photoreceptor cilium. Journal of Neuroscience, 19:6267-6274, 1999.
PMID: 10414956Link is external(Link is external)  PMCID: PMC6782817Link is external(Link is external) DOI: 10.1523/JNEUROSCI.19-15-06267.1999Link is external(Link is external)

Richardson GP, Forge A, Kros CJ, Marcotti W, Becker D, Williams DS, Thorpe J, Fleming J, Brown SDM, Steel KP: A missense mutation in myosin VIIA prevents aminoglycoside accumulation in cochlear hair cells. Annals of the New York Academy of Sciences, 884:110-124, 1999.
PMID: 10842588Link is external(Link is external)  PMCID:  DOI:

Wolfrum U, Liu X, Schmitt A, Udovichenko IP, Williams DS: Myosin VIIa as a common component of cilia and microvilli. Cell Motility and the Cytoskeleton, 40:261-271, 1998.
PMID: 9678669Link is external(Link is external)  PMCID:   DOI: 10.1002/(SICI)1097-0169(1998)40:33.0.CO;2-GLink is external(Link is external)

Liu X, Ondek B, Williams DS: Mutant myosin VIIa causes defective melanosome distribution in the RPE of shaker-1 mice. Nature Genetics, 19:117-118, 1998.
PMID: 9620764Link is external(Link is external)  PMCID:   DOI: 10.1038/470Link is external(Link is external)

Udovichenko IP, Newton AC, Williams DS: Regulation of the phosphorylation state of rhodopsin by dopamine. Journal of Biological Chemistry, 273:7181-7184, 1998.
PMID: 9516406Link is external(Link is external)  PMCID:   DOI: 10.1074/jbc.273.13.7181Link is external(Link is external)

Liu X, Vansant G, Udovichenko IP, Wolfrum U, Williams DS: Myosin VIIa, the product of the Usher 1B syndrome gene, is concentrated in the connecting cilia of rod and cone photoreceptor cells. Cell Motility and the Cytoskeleton, 37:240-252 (plus front cover), 1997.
PMID: 9227854Link is external(Link is external)  PMCID:   DOI: 10.1002/(SICI)1097-0169(1997)37:33.0.CO;2-ALink is external(Link is external)

Williams DS, Liu X, Schlamp CS, Ondek B, Jaken S, and Newton AC: Characterization of protein kinase C in photoreceptor outer segments. Journal of Neurochemistry, 69:1693-1702, 1997.
PMID: 9326298Link is external(Link is external)  PMCID:   DOI: 10.1046/j.1471-4159.1997.69041693.xLink is external(Link is external)

Boesze-Battaglia K, Kong F, Lamba OP, Stefano FP, Williams DS: Purification and light-dependent phosphorylation of a candidate fusion protein, the photoreceptor cell peripherin/rds.
Biochemistry, 36:6835-6846, 1997.
PMID: 9184167Link is external(Link is external)  PMCID:   DOI: 10.1021/bi9627370Link is external(Link is external)

Greene NM, Williams DS, Newton AC: Identification of protein kinase C phosphorylation sites on bovine rhodopsin. Journal of Biological Chemistry, 272:10341-10344, 1997.
PMID: 9099669Link is external(Link is external)  PMCID:   DOI: 10.1074/jbc.272.16.10341Link is external(Link is external)

Udovichenko IP, Newton AC, Williams DS: Contribution of protein kinase C to the phosphorylation of rhodopsin in intact retinas. Journal of Biological Chemistry, 272:7952-7976, 1997.
PMID: 9065465Link is external(Link is external)  PMCID:   DOI: 10.1074/jbc.272.12.7952Link is external(Link is external)

Liu X, Wu, T-H, Stowe, S, Atsuko M, Arikawa K, Naash M, Williams DS: Defective phototransductive disk membrane morphogenesis in transgenic mice expressing rhodopsin with a mutated N-terminal domain. Journal of Cell Science, 110:2589-2597, 1997.
PMID: 9372448Link is external(Link is external)  PMCID:   DOI:

Schlamp CS, Williams DS: Myosin V in the retina: localization in the rod photoreceptor synapse. Experimental Eye Research, 63:613-619, 1996.
PMID: 9068368Link is external(Link is external)  PMCID:  DOI: 10.1006/exer.1996.0155Link is external(Link is external)

Hicks JL, Liu X, Williams DS: The role of the ninaC proteins in photoreceptor cell structure: ultrastructure of ninaC deletion mutants and binding to actin filaments. Cell Motility and the Cytoskeleton, 35:367-379 (plus front cover), 1996.
PMID: 8956007Link is external(Link is external)  PMCID:   DOI: 10.1002/(SICI)1097-0169(1996)35:43.0.CO;2-3Link is external(Link is external)

Hallett MA, Delaat JL, Arikawa K, Schlamp CL, Kong F, Williams DS: Distribution of guanylate cyclase within rod photoreceptor outer segments. Journal of Cell Science, 109:1803-1812, 1996.
PMID: 8832403Link is external(Link is external)  PMCID:   DOI:

Azarian SM, King AJ, Hallett MA, Williams DS: Selective proteolysis of arrestin by calpain: molecular characterization and its effect on rhodopsin dephosphorylation. Journal of Biological Chemistry, 270:24375-24384, 1995.
PMID: 7592650Link is external(Link is external)  PMCID:   DOI: 10.1074/jbc.270.41.24375Link is external(Link is external)

Greene NM, Williams DS, Newton AC: Kinetics and localization of the phosphorylation of rhodopsin by protein kinase C. Journal of Biological Chemistry, 270:6710-6717, 1995.
PMID: 7896814Link is external(Link is external)  PMCID:   DOI: 10.1074/jbc.270.12.6710Link is external(Link is external)

Azarian SM, Williams DS: Calpain activity in the retinas of normal and RCS rats. Current Eye Research, 14:731-735, 1995.
PMID: 8529410Link is external(Link is external)  PMCID:   DOI: 10.3109/02713689508998502Link is external(Link is external)

Williams DS: The dynamics of cytosolic calcium in photoreceptor cells. Bioessays, 17: 282-286, 1995.
PMID: 7741721Link is external(Link is external)  PMCID:   DOI: 10.1002/bies.950170403Link is external(Link is external)

Williams DS, Park S, Schlamp, CL, Newton AC: Protein kinase C association with the retinal cytoskeleton and phosphorylation of vimentin. Experimental Eye Research, 58:747-759, 1994.
PMID: 7925714Link is external(Link is external)  PMCID:   DOI: 10.1006/exer.1994.1072Link is external(Link is external)

Nickells RW, Schlamp CL, Newton AC, Williams DS: Gene expression of the neuropeptide processing enzyme carboxypeptidase E in rat photoreceptor cells. Journal of Neurochemistry, 61:1891-1900, 1993.
PMID: 7693871Link is external(Link is external)  PMCID:   DOI: 10.1111/j.1471-4159.1993.tb09831.xLink is external(Link is external)

Nickells RW, Newton AC, Williams DS: PCR reamplification of small target cDNAs from high-resolution polyacrylamide gels. Amplifications, 9:6-7, 1993.
PMID:   PMCID:   DOI:

Newton AC, Williams DS: Rhodopsin is the major in situ substrate of protein kinase C in rod outer segments of photoreceptors. Journal of Biological Chemistry, 268:18181-18186, 1993.
PMID: 8349693Link is external(Link is external)  PMCID:   DOI:

Newton AC, Williams DS: Does protein kinase C play a role in rhodopsin desensitization? Trends in Biochemical Sciences, 18:275-277, 1993.
PMID: 8236436Link is external(Link is external)  PMCID:   DOI: 10.1016/0968-0004(93)90032-iLink is external(Link is external)

Azarian SM, Schlamp CL, Williams DS: Characterization of calpain II in the retina and photoreceptor outer segments. Journal of Cell Science, 105:787-798, 1993.
PMID: 8408304Link is external(Link is external)  PMCID:  DOI:

Arikawa K, Williams DS: Acetylated alpha-tubulin in the connecting cilium of developing rat photoreceptors. Investigative Ophthalmology and Visual Science, 34:2145-2149, 1993.
PMID: 8491566Link is external(Link is external)  PMCID:   DOI:

Porter JA, Hicks JL, Willliams DS, Montell C: Differential localizations of and requirements for the two Drosophila ninaC kinase/myosins in photoreceptor cells. Journal of Cell Biology, 116:683-693, 1992.
PMID: 1730774Link is external(Link is external)  PMCID: PMC2289314Link is external(Link is external)  DOI: 10.1083/jcb.116.3.683Link is external(Link is external)

Hicks JL, Williams DS: Distribution of the myosin I-like ninaC proteins in the Drosophila retina and ultrastructural analysis of mutant phenotypes. Journal of Cell Science, 101:247-254, 1992.
PMID: 1569127Link is external(Link is external)  PMCID:   DOI:

Arikawa K, Molday LL, Molday RS, Williams DS: Localization of peripherin/rds in the disk membranes of cone and rod photoreceptors: relationship to disk membrane morphogenesis and retinal degeneration. Journal of Cell Biology, 116:659-667, 1992.
PMID: 1730772Link is external(Link is external)  PMCID: PMC2289304Link is external(Link is external)  DOI: 10.1083/jcb.116.3.659Link is external(Link is external)

Williams DS, Hallett M, Arikawa K: Association of myosin with the connecting cilium of rod photoreceptors. Journal of Cell Science, 103:183-190, 1992.
PMID: 1429904Link is external(Link is external)  PMCID:   DOI:

Williams DS, Roberts EA: Modification of the daily photoreceptor membrane shedding response in vitro by antioxidants. Investigative Ophthalmology and Visual Science, 33:3005-3008, 1992.
PMID: 1526748Link is external(Link is external)  PMCID:   DOI:

Päällysaho T, Williams DS: Epithelial cell-substrate adhesion in the cornea: localization of actin, talin, integrin and fibronectin. Experimental Eye Research, 52:261-267, 1991.
PMID: 1901797Link is external(Link is external)  PMCID:   DOI: 10.1016/0014-4835(91)90089-wLink is external(Link is external)

Newton AC, Williams DS: Involvement of protein kinase C in the phosphorylation of rhodopsin. Journal of Biological Chemistry, 266:17725-17728, 1991.
PMID: 1917916Link is external(Link is external)  PMCID:   DOI:

Arikawa K, Williams DS: Alpha-actinin and actin in the outer retina: a double immunoelectron microscopical study. Cell Motility and the Cytoskeleton, 18:15-25 (plus front cover), 1991.
PMID: 2004431Link is external(Link is external)  PMCID:   DOI: 10.1002/cm.970180103Link is external(Link is external)

Williams DS: Actin filaments and photoreceptor membrane turnover. Bioessays, 13:171-178, 1991.
PMID: 1859395Link is external(Link is external)  PMCID:   DOI: 10.1002/bies.950130405Link is external(Link is external)

Arikawa K, Hicks JL, Williams DS: Identification of actin filaments in the rhabdomeral microvilli of Drosophila photoreceptors. Journal of Cell Biology, 110:1993-1998, 1990.
PMID: 2112548Link is external(Link is external)  PMCID: PMC2116135Link is external(Link is external)  DOI: 10.1083/jcb.110.6.1993Link is external(Link is external)

Williams DS, Arikawa K, Päällysaho T: Cytoskeletal components of the adherens junctions between the photoreceptors and the supportive Müller cells. Journal of Comparative Neurology, 295:155-164, 1990.
PMID: 2341633Link is external(Link is external)  PMCID:   DOI: 10.1002/cne.902950113Link is external(Link is external)

Williams DS, Linberg KA, Fisher SK: Shedding of cone photoreceptor disk membranes in lizard eyecups in vitro. Experimental Eye Research, 50:231-234, 1990.
PMID: 2311685Link is external(Link is external)  PMCID:   DOI: 10.1016/0014-4835(90)90236-nLink is external(Link is external)

Arikawa K, Williams DS: Organization of actin filaments and immunocolocalization of alpha-actinin in the connecting cilium of rat photoreceptors. Journal of Comparative Neurology, 288:640-646, 1989.
PMID: 2808754Link is external(Link is external)  PMCID:   DOI: 10.1002/cne.902880410Link is external(Link is external)

Williams DS, Shuster TA, Moldrawski MR, Blest AD, Farber DB: Isolation of rod outer segments on Percoll gradients: effect of specific protease inhibition. Experimental Eye Research, 49:439-444, 1989.
PMID: 2551708Link is external(Link is external)  PMCID:   DOI: 10.1016/0014-4835(89)90052-3Link is external(Link is external)

Anderson DH, Williams DS, Neitz J, Farris RN, Fliesler SJ: Tunicamycin-induced degeneration in cone photoreceptors. Visual Neuroscience, 1:153-158, 1988.
PMID: 3154792Link is external(Link is external)  PMCID:   DOI: 10.1017/s0952523800001425Link is external(Link is external)

Williams DS, Linberg KA, Vaughan DK, Fariss RN, Fisher SK: Disruption of microfilament organization and deregulation of disk membrane morphogenesis by cytochalasin D in rod and cone photoreceptors. Journal of Comparative Neurology, 272:161-176, 1988. 
PMID: 3397406Link is external(Link is external)  PMCID:   DOI: 10.1002/cne.902720202Link is external(Link is external)

Shuster TS, Walter AE, Williams DS, Farber DB: Identification of a PNA-binding protein from human retina. Experimental Eye Research, 45:685-694, 1987.
PMID: 3428393Link is external(Link is external)  PMCID:   DOI: 10.1016/s0014-4835(87)80117-3Link is external(Link is external)

Williams DS, Colley NJ, Farber DB: Photoreceptor degeneration in a pure-cone retina: effects of cyclic nucleotides, and inhibitors of phosphodiesterase and protein synthesis. Investigative Ophthalmology and Visual Science, 28:1059-1069, 1987.
PMID: 2439472Link is external(Link is external)  PMCID:   DOI:

Williams DS, Fisher SK: Prevention of the shedding of rod outer segment disks by detachment from the retinal pigment epithelium. Investigative Ophthalmology and Visual Science, 28:184-187, 1987.
PMID: 3804649Link is external(Link is external)  PMCID:   DOI:

Williams DS, Colley NJ, Anderson DH, Farber DB, Fisher SK: In vitro maintenance of a pure-cone retina. Investigative Ophthalmology and Visual Science, 27:666-673, 1986.
PMID: 3516917Link is external(Link is external)  PMCID:   DOI:

Williams DS, Wilson C, Linberg K, Fisher SK: Effects of low sodium, ouabain, and strophanthidin on the shedding of rod outer segment discs. Journal of Comparative Physiology A, 155:763-770, 1984.
PMID:   PMCID:   DOI: 
http://link.springer.com/content/pdf/10.1007/BF00611593.pdfLink is external(Link is external)

Williams DS: Changes in photoreceptor performance associated with the daily turnover of photoreceptor membrane in locusts. Journal of Comparative Physiology A, 150:509-519, 1983.
PMID:   PMCID:   DOI:
http://link.springer.com/content/pdf/10.1007/BF00609577.pdfLink is external(Link is external)

Blest AD, Stowe S, Eddey W, Williams DS: The local deletion of a microvillar cytoskeleton from photoreceptors of tipulid flies during membrane turnover. Proceedings of the Royal Society of London B, 215:469-489, 1982.
PMID: 6127716Link is external(Link is external)  PMCID:   DOI: 10.1098/rspb.1982.0054Link is external(Link is external)

Williams DS: Rhabdom size and photoreceptor membrane turnover in a muscoid fly. Cell and Tissue Research, 226:629-639, 1982.
PMID: 7139695Link is external(Link is external)  PMCID:   DOI: 10.1007/BF00214790Link is external(Link is external)

Williams DS: Photoreceptor membrane shedding and assembly can be initiated locally within an insect eye. Science, 218:898-900 (plus front cover), 1982.
PMID: 7134980Link is external(Link is external)  PMCID:   DOI: 10.1126/science.7134980Link is external(Link is external)

Williams DS: Ommatidial structure in relation to photoreceptor membrane turnover in a locust. Cell and Tissue Research, 225:595-617, 1982.
PMID: 7127410Link is external(Link is external)  PMCID:   DOI: 10.1007/BF00214807Link is external(Link is external)

Blest AD, Hardie RC, McIntyre P, Williams DS: The spectral sensitivities of identified receptors and the function of retinal tiering in the principal eyes of a jumping spider. Journal of Comparative Physiology A, 145:227-239, 1981.
PMID:   PMCID:   DOI:
http://link.springer.com/content/pdf/10.1007/BF00605035.pdfLink is external(Link is external)

Williams DS: Twisted rhabdomeres in the compound eye of the tipulid fly (Diptera). Cell and Tissue Research, 217:625-632, 1981.
PMID: 7249053Link is external(Link is external)  PMCID:   DOI: 10.1007/BF00219369Link is external(Link is external)

Williams DS: A jumping spider's view. Hemisphere, 26:178-183, 1981.
PMID:   PMCID:   DOI:

Blest AD, Williams DS, Kao L: The posterior median eyes of the dinopid spider Menneus. Cell and Tissue Research, 211:391-403, 1980.
PMID: 7417991Link is external(Link is external)  PMCID:   DOI: 10.1007/BF00234395Link is external(Link is external)

Williams DS, McIntyre P: The principal eyes of a jumping spider have a telephoto component. Nature, 288:578-580, 1980.
PMID:   PMCID:   DOI: 10.1038/288578a0Link is external(Link is external)

Williams DS, Blest AD: Extracellular shedding of photoreceptor membrane in the open rhabdom of a tipulid fly. Cell and Tissue Research, 205:423-438, 1980.
PMID: 7357581Link is external(Link is external)  PMCID:   DOI: 10.1007/BF00232283Link is external(Link is external)

Williams DS: Organisation of the compound eye of a tipulid fly during the day and night. Zoomorphologie, 95:85-104, 1980.
PMID:   PMCID:   DOI:
http://link.springer.com/content/pdf/10.1007/BF00997903.pdfLink is external(Link is external)

Williams DS: Ca++-induced structural changes in photoreceptor microvilli of Diptera. Cell and Tissue Research, 206:225-232, 1980.
PMID: 6771011Link is external(Link is external)  PMCID:   DOI: 10.1007/BF00232766Link is external(Link is external)

Williams DS: The feeding behaviour of New Zealand Dolomedes species (Aranaea; Pisauridae). New Zealand Journal of Zoology, 6:95-105, 1979.
PMID:   PMCID:   DOI: 10.1080/03014223.1979.10428352Link is external(Link is external)

Williams DS: The physiological optics of a nocturnal semi-aquatic spider, Dolomedes aquaticus (Pisauridae). Zeitschrift fuer Naturforschung, 34c:463-469, 1979.
PMID:   PMCID:   DOI: 10.1515/znc-1979-5-625Link is external(Link is external)

Books, Book Chapters, and Special Editions

Umapathy A, Williams DS: Live Imaging of Organelle Motility in RPE Flatmounts.  Advances in Experimental Medicine and Biology, 1185:389-393, 2019.
PMID: 31884643Link is external(Link is external)  PMCID:   DOI: 10.1007/978-3-030-27378-1_64Link is external(Link is external)

Volland S, Williams DS: Preservation of photoreceptor nanostructure for electron tomography using transcardiac perfusion followed by high-pressure freezing and freeze-substitution.  Advances in Experimental Medicine and Biology, 1074:603-607, 2018.
PMID:  29721993Link is external(Link is external)  PMCID: PMC6501585Link is external(Link is external)   DOI: 10.1007/978-3-319-75402-4_73Link is external(Link is external) 

Hazim RA, Williams DS: Cell culture analysis of the phagocytosis of photoreceptor outer segments by primary mouse RPE cells.  In: Tanimoto N. (eds) Mouse Retinal Phenotyping. Methods in Molecular Biology, 1753:63-71. Humana Press, New York, NY, 2018.
PMID:  29564781Link is external(Link is external)  PMCID: PMC6531314Link is external(Link is external)  DOI: 10.1007/978-1-4939-7720-8_4Link is external(Link is external)

Hazim RA, Jiang M, Esteve-Rudd J, Diemer T, Lopes VS, Williams DS:  Live-cell imaging of phagosome motility in primary mouse RPE cells. Advances in Experimental Medicine and Biology, 854:751-755, 2016.
PMID: 26427485Link is external(Link is external)  PMCID:   DOI: 10.1007/978-3-319-17121-0_100Link is external(Link is external) 

Lopes VS, Williams DS: Gene therapy for the retinal degeneration of Usher syndrome due to mutations in MYO7A. Retinal Disorders Genetic Approaches to Diagnosis and Treatment. Cold Spring Harbor Perspectives in Medicine, 5(6), 2015.
PMID: 25605753Link is external(Link is external)  PMCID:   DOI: 10.1101/cshperspect.a017319Link is external(Link is external)

Sernagor E, Williams DS: Treatment strategies for retinal dystrophies. Guest Editors. Visual Neuroscience, 31:287-288, 2014.
PMID: 25346971Link is external(Link is external)  PMCID:   DOI: 10.1017/s0952523814000273Link is external(Link is external)

Lopes VS, Diemer T, Williams DS: Assessment of different virus-mediated approaches for retinal gene therapy of Usher 1B. Advances in Experimental Medicine and Biology, 801:725-31, 2014.
PMID: 24664764Link is external(Link is external)  PMCID:   DOI: 10.1007/978-1-4614-3209-8_91Link is external(Link is external)

Esteve-Rudd J, Lopes VS, Jiang M, Williams DS: In vivo and in vitro monitoring of phagosome maturation in retinal pigment epithelium cells. Advances in Experimental Medicine and Biology, 801:85-90, 2014.
PMID: 24664684Link is external(Link is external)  PMCID:   DOI: 10.1007/978-1-4614-3209-8_11Link is external(Link is external)

Williams DS, Lopes VS: Gene Therapy Strategies for Usher Syndrome Type 1B. Advances in Experimental Medicine and Biology, 723:235-42, 2012.
PMID: 22183338Link is external(Link is external)  PMCID:   DOI: 10.1007/978-1-4614-0631-0_31Link is external(Link is external)

Trivedi D, Williams DS: Ciliary transport of opsin. Advances in Experimental Medicine and Biology, 664:185-191, 2010.
PMID: 20238016Link is external(Link is external)  PMCID: PMC2933454Link is external(Link is external)  DOI: 10.1007/978-1-4419-1399-9_21Link is external(Link is external)

Williams DS, Anderson DH: Rod and cone photoreceptor cells: outer segment membrane renewal. In: Encyclopedia of the Eye, Vol 4. Ed: Dartt, DA. Academic Press, Oxford, UK, pp. 143-146, 2010.
PMID:  PMCID:   DOI: 10.1016/B978-0-12-374203-2.00175-5Link is external(Link is external)

Anderson DH, Williams DS: Rod and cone photoreceptor cells: inner and outer segments. In: Encyclopedia of the Eye, Vol 4. Ed: Dartt, DA. Academic Press, Oxford, UK, pp. 139-142, 2010.
PMID:   PMCID:   DOI: 10.1016/B978-0-12-374203-2.00173-1Link is external(Link is external)

Diemer T, Gibbs D, Williams DS: Analysis of the rate of disk membrane digestion by cultured RPE cells. Advances in Experimental Medicine and Biology, 613:321-326, 2008.
PMID:   PMCID:   DOI: 10.1007/978-0-387-74904-4_37Link is external(Link is external) 

Williams DS: Retinal degeneration in Usher syndrome. In: Retinal Degenerations: Biology, Diagnostics, and Therapeutics. Eds: Barnstable C, Tombran-Tink J. Humana Press, New Jersey, pp. 137-148, 2007.
PMID:   PMCID:   DOI: 10.1007/978-1-59745-186-4_7Link is external(Link is external) 

Lillo C, Kitamoto J, Williams DS: Roles and interactions of Usher 1 proteins in the outer retina. Advances in Experimental Medicine and Biology, 572:341-348, 2006.
PMID:   PMCID:   DOI: 10.1007/0-387-32442-9_48Link is external(Link is external) 

Williams DS, editor: Photoreceptor Cell Biology and Inherited Retinal Degenerations. World Scientific Publishing, Singapore, 2004.
PMID:   PMCID:   DOI: doi.org/10.1142/5557Link is external(Link is external) 

Williams DS, Gibbs D: Myosin VIIa in the retina. In: Photoreceptor Cell Biology and Inherited Retinal Degenerations. Ed: Williams DS. World Scientific Publishing, Singapore, pp. 397-436, 2004.
PMID:   PMCID:   DOI: 10.1142/9789812561756_0015Link is external(Link is external) 

Williams DS: Cell biology of photoreceptors and the RPE: A brief introduction. In: Photoreceptor Cell Biology and Inherited Retinal Degenerations. Ed: Williams DS. World Scientific Publishing, Singapore, pp. 1-6, 2004.
PMID:   PMCID:   DOI: 10.1142/9789812561756_0001Link is external(Link is external) 

Gibbs D, Williams DS: Isolation and culture of primary mouse retinal pigmented epithelial cells. Advances in Experimental Medicine and Biology, 533:347-352, 2003.
PMID:   PMCID:   DOI: 10.1007/978-1-4615-0067-4_44Link is external(Link is external)

Slatter T, Azarian SM, Tebbutt S, Maw M, Williams DS: Screen for Usher syndrome 1B mutations in the ovine myosin VIIa gene. Advances in Experimental Medicine and Biology, 533:151-155, 2003.
PMID:   PMCID:   DOI: 10.1007/978-1-4615-0067-4_19Link is external(Link is external) 

Lillo C, Kitamoto J, Liu X, Quint E, Steel KP, Williams DS: Mouse models for Usher syndrome 1B. Advances in Experimental Medicine and Biology, 533:143-150, 2003.
PMID:   PMCID:   DOI: 10.1007/978-1-4615-0067-4_18Link is external(Link is external) 

Williams DS, Liu X, Vansant G, Ondek, B: Blindness in Usher syndrome 1B: myosin VIIa in the retina. In: Retinal Degenerative Diseases and Experimental Therapy. Eds: Hollyfield JG, Anderson RE, and LaVail MM, Plenum Press, New York, pp 15-26, 1999.
PMID:   PMCID:   DOI: 10.1007/978-0-585-33172-0_2Link is external(Link is external)

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