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Prepublished online as a Blood First Edition Paper on August 22, 2002; DOI 10.1182/blood-2002-04-1169. This Article Full Text Full Text (PDF) All Versions of this Article: 2002-04-1169v1 100/13/4655    most recent Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Rights and Permissions Citing Articles Citing Articles via HighWire Citing Articles via CrossRef Citing Articles via Google Scholar Google Scholar Articles by Donovan, A. Articles by Zon, L. I. Search for Related Content PubMed PubMed Citation Articles by Donovan, A. Articles by Zon, L. I. Related Collections Red Cells Social Bookmarking                   What's this?  Previous Article  |  Table of Contents  |  Next Article  Blood, 15 December 2002, Vol. 100, No. 13, pp. 4655-4659 RED CELLS The zebrafish mutant gene chardonnay (cdy) encodes divalent metal transporter 1 (DMT1) Adriana Donovan, Alison Brownlie, Michael O. Dorschner, Yi Zhou, Stephen J. Pratt, Barry H. Paw, Ruth B. Phillips, Christine Thisse, Bernard Thisse, and Leonard I. Zon From the Department of Hematology/Oncology, and Howard Hughes Medical Institute, Children's Hospital, Boston MA; Institut de Génétique et de Biologie Moléculaire et Cellulaire, CNRS/Institut National de la Santé et de la Recherche Médicale (INSERM)/ULP, BP 163, CU de Strasbourg 1, BP163, 67404 Illkirch Cedex, France; Department of Medicine, University of Washington, Seattle; School of Biological Sciences, Washington State University, Vancouver; Department of Medicine, Washington University, St Louis MO; and Xenon Genetics, Burnaby, BC, Canada. Iron is an essential nutrient required for the function of all cells, most notably for the production of hemoglobin in red blood cells. Defects in the mechanisms of iron absorption, storage, or utilization can lead to disorders of iron-limited erythropoiesis or iron overload. In an effort to further understand these processes, we have used the zebrafish as a genetic system to study vertebrate iron metabolism. Here we characterized the phenotype of chardonnay (cdy), a zebrafish mutant with hypochromic, microcytic anemia, and positioned the mutant gene on linkage group 11. The cdy gene was isolated by a functional genomics approach in which we used a combination of expression studies, sequence analyses, and radiation hybrid panel mapping. We identified erythroid-specific genes using a whole embryo mRNA in situ hybridization screen and placed these genes on the zebrafish genomic map. One of these genes encoded the iron transporter divalent metal transporter 1 (DMT1) and colocalized with the cdy gene. We identified a nonsense mutation in the cdy allele and demonstrated that, whereas wild-type zebrafish DMT1 protein can transport iron, the truncated protein expressed in cdy mutants is not functional. Our studies further demonstrate the conservation of iron metabolism in vertebrates and suggest the existence of an alternative pathway of intestinal and red blood cell iron uptake. © 2002 by The American Society of Hematology.   CiteULike    Connotea    Del.icio.us    Digg    Reddit    Technorati    What's this? This article has been cited by other articles: N. C. Andrews Forging a field: the golden age of iron biology Blood, July 15, 2008; 112(2): 219 - 230. [Full Text] [PDF] I. De Domenico, M. B. Vaughn, D. Yoon, J. P. Kushner, D. M. Ward, and J. Kaplan Zebrafish as a model for defining the functional impact of mammalian ferroportin mutations Blood, November 15, 2007; 110(10): 3780 - 3783. [Abstract] [Full Text] [PDF] A. L. Lumsden, T. L. Henshall, S. Dayan, M. T. Lardelli, and R. I. Richards Huntingtin-deficient zebrafish exhibit defects in iron utilization and development Hum. Mol. Genet., August 15, 2007; 16(16): 1905 - 1920. [Abstract] [Full Text] [PDF] A. Donovan, C. N. 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