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Institute of Neuroscience Faculty

Monte Westerfield

Professor, Department of Biology
A.B., 1973, Princeton University
Ph.D., 1977, Duke University

Research Interests

Ear and eye development and pathogenesis; molecular genetic analysis of neuronal development
Laboratory Website
Zebrafish Database (ZFIN)

Our laboratory focuses on understanding the mechanisms that regulate the differentiation of neurons.  We study how the specific properties of different kinds of neurons are regulated. 

We use zebrafish and a combination of anatomical, physiological, molecular, and genetic techniques.  The goal of our research is to provide a better understanding of the mechanisms that regulate the establishment of specific neuronal cell fates during normal development and what goes wrong during disease. 

Current research focuses on models of human disease. In particular, we are making and studying models of Usher syndrome, the leading cause of deafblindness.

Our research is funded by the National Eye Institute, the National Institute on Deafness and Other Communicative Disorders, the National Institute of Child Health and Development, and the Office of the Director of the National Institutes of Health. We also thank friends and family of Jodi Carter and Ryan Thomason and The Megan Foundation for generous donations to the Usher Syndrome Research Fund that supports our work.

Contribute to the Usher Syndrome Research Fund.


Deans, A. R., S. E. Lewis, E. Huala, S. S. Anzaldo, M. Ashburner, J. P. Balhoff, D. C. Blackburn, et al.  (2015).  Finding Our Way through Phenotypes.  PLoS Biol, 13(1), e1002033. PMC25562316.

Elsayed, S. M., J. B. Phillips, R. Heller, M. Thoenes, E. Elsobky, G. Nurnberg, P. Nurnberg, et al.  (2015).  Non-manifesting AHI1 truncations indicate localized loss-of-function tolerance in a severe Mendelian disease gene.  Hum Mol Genet. (in press).

Beck, B. B., J. B. Phillips, M. P. Bartram, J. Wegner, M. Thoenes, A. Pannes, J. Sampson, et al.  (2014).  Mutation of POC1B in a Severe Syndromic Retinal Ciliopathy.  Hum Mutat, 35(10), 1153-62. PMC25044745.

Blanco-Sánchez, B., A. Clement, J. Fierro, Jr., P. Washbourne, and M. Westerfield.  (2014).  Complexes of Usher proteins preassemble at the endoplasmic reticulum and are required for trafficking and ER homeostasis.  Dis Model Mech, 7(5), 547-59. PMC4007406.

Köhler, S., S. C. Doelken, C. J. Mungall, S. Bauer, H. V. Firth, I. Bailleul-Forestier, G. C. Black, et al.  (2014).  The Human Phenotype Ontology project: linking molecular biology and disease through phenotype data.  Nucleic Acids Res, 42(Database issue), D966-74. PMC24217912.

Phillips, J. B. and M. Westerfield.  (2014).  Zebrafish models in translational research: tipping the scales toward advancements in human health.  Dis Model Mech, 7(7), 739-743. PMC24973743.

Van Slyke, C. E., Y. M. Bradford, M. Westerfield, and M. A. Haendel.  (2014).  The zebrafish anatomy and stage ontologies: representing the anatomy and development of Danio rerio.  J Biomed Semantics, 5(1), 12. PMC3944782.

Consortium.  (2013).  Gene Ontology annotations and resources.  Nucleic Acids Res, 41(Database issue), D530-5. PMC3531070.

Doelken, S. C., S. Kohler, C. J. Mungall, G. V. Gkoutos, B. J. Ruef, C. Smith, D. Smedley, et al.  (2013).  Phenotypic overlap in the contribution of individual genes to CNV pathogenicity revealed by cross-species computational analysis of single-gene mutations in humans, mice and zebrafish.  Dis Model Mech, 6, 358-372. PMC3597018.

Howe, D. G., Y. M. Bradford, T. Conlin, A. E. Eagle, D. Fashena, K. Frazer, J. Knight, et al.  (2013).  ZFIN, the Zebrafish Model Organism Database: increased support for mutants and transgenics.  Nucleic Acids Res, 41(D1), D854-60. PMC3531097.

Howe, K., M. D. Clark, C. F. Torroja, J. Torrance, C. Berthelot, M. Muffato, J. E. Collins, et al.  (2013).  The zebrafish reference genome sequence and its relationship to the human genome.  Nature, 496, 498-503. PMC3703927.

Köhler, S., S. C. Doelken, B. J. Ruef, S. Bauer, N. Washington, M. Westerfield, G. Gkoutos, et al.  (2013).  Construction and accessibility of a cross-species phenotype ontology along with gene annotations for biomedical research.  F1000 Research, 2, 30. PMC3799545.

Midford, P. E., T. A. Dececchi, J. P. Balhoff, W. M. Dahdul, N. Ibrahim, H. Lapp, J. G. Lundberg, et al.  (2013).  The vertebrate taxonomy ontology: a framework for reasoning across model organism and species phenotypes.  J Biomed Semantics, 4(1), 34. PMC24267744.

Phillips, J. B., H. Vastinsalo, J. Wegner, A. Clement, E. M. Sankila, and M. Westerfield.  (2013).  The cone-dominant retina and the inner ear of zebrafish express the ortholog of CLRN1, the causative gene of human Usher syndrome type 3A.  Gene Expr Patterns, 13(8), 473-481. PMC3888827.

Smedley, D., A. Oellrich, S. Kohler, B. Ruef, M. Westerfield, P. Robinson, S. Lewis, and C. Mungall.  (2013).  PhenoDigm: analyzing curated annotations to associate animal models with human diseases.  Database, 2013, bat025. PMC3649640.

Sullivan, J., K. Karra, S. A. Moxon, A. Vallejos, H. Motenko, J. D. Wong, J. Aleksic, et al.  (2013).  InterMOD: integrated data and tools for the unification of model organism research.  Scientific reports, 3, 1802. PMC3647165.

Chen, C. K., C. J. Mungall, G. V. Gkoutos, S. C. Doelken, S. Kohler, B. J. Ruef, C. Smith, et al.  (2012).  MouseFinder: Candidate disease genes from mouse phenotype data.  Hum Mutat, 33(5), 858-66. PMC3327758.

Consortium.  (2012).  The Gene Ontology: enhancements for 2011.  Nuc Acids Res, 40(Database issue), D559-64. PMC3245151.

Dahdul, W. M., J. P. Balhoff, D. C. Blackburn, A. D. Diehl, M. A. Haendel, B. K. Hall, H. Lapp, et al.  (2012).  A unified anatomy ontology of the vertebrate skeletal system.  PLoS One, 7(12), e51070. PMC3519498.

Gao, J., C. Zhang, B. Yang, L. Sun, M. Westerfield, and G. Peng.  (2012).  Dcc Regulates Asymmetric Outgrowth of Forebrain Neurons in Zebrafish.  PLoS One, 7(5), e36516. PMC3351449.

Mabee, P., J. Balhoff, W. Dahdul, H. Lapp, P. Midford, T. Vision, and M. Westerfield.  (2012).  500,000 fish phenotypes: The new informatics landscape for evolutionary and developmental biology of the vertebrate skeleton.  J Appl Ichthyol, 28, 300-305. PMC22736877.

Bradford, Y., T. Conlin, N. Dunn, D. Fashena, K. Frazer, D.G. Howe, J. Knight, P. Mani, R. Martin, S.A. Moxon, H. Paddock, C. Pich, S. Ramachandran, B.J. Ruef, L. Ruzicka, H. Bauer Schaper, K. Schaper, X. Shao, A. Singer, J. Sprague, B. Sprunger, C. Van Slyke, and M. Westerfield. (2011). ZFIN: enhancements and updates to the Zebrafish Model Organism Database. Nucleic Acids Res, 39(Database issue), D822-9.

Detrich, H.W., 3rd, M. Westerfield, and L.I. Zon. (2011). The zebrafish: cellular and developmental biology, part B. Methods Cell Biol, 101.

Detrich, H. W., 3rd, M. Westerfield, and L. I. Zon, The Zebrafish: Genetics, Genomics and Informatics. 3 ed. Meth Cell Biol. Vol. 104. 2011, San Diego CA: Academic Press.

Detrich, H. W., 3rd, M. Westerfield, and L. I. Zon, The Zebrafish: Disease Models and Chemical Screens. 3 ed. Meth Cell Biol. Vol. 105. 2011, San Diego, CA: Academic Press.

Detrich, H. W., 3rd, M. Westerfield, and L. I. Zon, The Zebrafish: Genetics, Genomics and Informatics. Preface. 2011/09/20 ed. Meth Cell Biol. Vol. 104. 2011, San Diego: Academic Press. xix-xx.

Howe, D. G., K. Frazer, D. Fashena, L. Ruzicka, Y. Bradford, S. Ramachandran, B. J. Ruef, et al.  (2011).  Data Extraction, Transformation, and Dissemination through ZFIN.  Meth Cell Biol, 104, 311-25.

Murray KN, Bauer J, Tallen A, Matthews JL, Westerfield M, Varga ZM. (2011). Characterization and management of asymptomatic Mycobacterium infections at the Zebrafish International Resource Center. Comp. Med. (in press).

Murray KN, Dreska M, Nasiadka A, Rinne M, Matthews JL, Carmichael C, Bauer J, Varga ZM, Westerfield M. (2011). Transmission, diagnosis, and recommendations for control of Pseudoloma neurophilia infections in laboratory zebrafish (Danio rerio) facilities. Comp. Med. (in press).

Phillips, J.B., B. Blanco-Sanchez, J.J. Lentz, A. Tallafuss, K. Khanobdee, S. Sampath, Z.G. Jacobs, P.F. Han, M. Mishra, D.S. Williams, B.J. Keats, P. Washbourne, and M. Westerfield. (2011). Harmonin (ush1c) is required in Müller glial cells for photoreceptor synaptic development and function in the zebrafish. Dis. Models Mech. 4, 000-000 doi:10.1242/dmm.006429.

DeLaurier, A., B.F. Eames, B. Blanco-Sanchez, G. Peng, X. He, M.E. Swartz, B. Ullmann, M. Westerfield, and C.B. Kimmel. (2010). Zebrafish sp7:EGFP: a transgenic for studying otic vesicle formation, skeletogenesis, and bone regeneration. Genesis, 48(8), 505-11.

Detrich, H.W., 3rd, M. Westerfield, and L.I. Zon. (2010). The Zebrafish: Cellular and Developmental Biology, Part A. Methods Cell Biol, 100. San Diego, CA: Academic Press. 347.

Detrich, H.W., 3rd, M. Westerfield, and L.I. Zon. (2010). The Zebrafish: Cellular and Developmental Biology, Part A. Preface. Methods Cell Biol, 100, xiii.

Ebermann, I., J.B. Phillips, M.C. Liebau, R.K. Koenekoop, B. Schermer, I. Lopez, E. Schafer, A.F. Roux, C. Dafinger, A. Bernd, E. Zrenner, M. Claustres, B. Blanco, G. Nurnberg, P. Nurnberg, R. Ruland, M. Westerfield, T. Benzing, and H.J. Bolz. (2010). PDZD7 is a modifier of retinal disease and a contributor to digenic Usher syndrome. J Clin Invest, 120(6), 1812-23.

Zhang, C., Y. Song, D.A. Thompson, M.A. Madonna, G.L. Millhauser, S. Toro, Z. Varga, M. Westerfield, J. Gamse, W. Chen, and R.D. Cone. (2010). Pineal-specific agouti protein regulates teleost background adaptation. Proc Natl Acad Sci U S A, 107(47), 20164-71.

Dahdul, W.M., Balhoff, J.P., Engeman, J., Grande, T., Hilton, E.J., Kothari, C., Lapp, H., Lundberg, J.G., Midford, P.E., Vision, T.J., Westerfield, M., and Mabee, P.M. (2010) Evolutionary characters, phenotypes and ontologies: curating data from the systematic biology literature. PLoS One. 5(5):e10708 PDF

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Last Updated 9/19/2014 -