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David Williams, Ph.D.

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Department / Division Affiliations
Professor, Neurobiology
Professor in Residence, Ophthalmology, Neuroscience IDP
Member, Access Biochemistry, Biophysics, Structural Biology Home Area, ACCESS Program: Neuroscience IDP, Access Cell and Developmental Biology Home Area, Access Neuroscience Home Area, Brain Research Institute, Jules Stein Eye Institute
Faculty, ACCESS Program

Research Interest:

Retinal cell biology

Dr. Williams' laboratory is interested primarily in two topics of retinal cell biology. In one, we are studying the photoreceptor cilium, which is extraordinarily modified to form the photoreceptor outer segment, and is affected by many ciliopathies. Projects include ciliogenesis, axonemal transport, barrier/filter functions, and 3-D ultrastructure. The second topic concerns the motor proteins involved in the motility of organelles in the retinal pigmented epithelium (RPE). We use a combination of mouse genetics, biochemistry, and different forms of microscopy, including live-cell imaging. We are also developing a gene therapy approach for the retinal degeneration associated with a form of Usher syndrome (inherited deaf-blindness, caused by mutations in an unconventional myosin gene), and are interested in relationships between retinal degenerations and other neurodegenerative disorders.


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. 2013; .
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 . 2012; 287: 11469-11480.
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. 2012; 29: 83-93.
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. 2012; 32: 10587-10593.
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 . 2011; 4: 786-800.
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. 2011; 52: 7924-7936.
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. 2011; 20: 2560-2570.
Williams DS, Lopes VS The many different cellular functions of MYO7A in the retina. Biochemical Society transactions. 2011; 39: 1207-10.
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 Al, 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 . 2010; 42: 175-180.
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. 2010; 21: 4076-4088.
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. 2010; 51: 1130-1135.
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 . 2010; 19: 4229-4238.
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. 2009; 29: 15810-15818.