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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 (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 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 Social Bookmarking                   What's this? Submitted April 17, 2002 Accepted July 21, 2002 The zebrafish mutant gene chardonnay encodes Divalent Metal Transporter1 (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* Department of Hematology/Oncology, Children's Hospital, Boston, MA, USA Xenon Genetics, Burnaby, BC, Canada Department of Medicine, University of Washington, Seattle, WA, USA Department of Medicine, Washington University, St. Louis, MO, USA School of Biological Sciences, Washington State University, Vancouver, WA, USA Institut de Genetique et de Biologie Moleculaire et Cellulaire, CNRS/INSERM/ULP, Illkirch, France Department of Hematology/Oncology, Children's Hospital, Boston, MA, USA; Howard Hughes Medical Institute, Children's Hospital, Boston, MA, USA * Corresponding author; email: zon{at}enders.tch.harvard.edu. 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 and/or utilization can lead to disorders of iron-limited erythropoiesis or iron overload. In an effort to further understand these processes we have employed the zebrafish as a genetic system to study vertebrate iron metabolism. Here we characterized the phenotype of chardonnay, a zebrafish mutant with hypochromic, microcytic anemia, and positioned the mutant gene on linkage group 11. The chardonnay gene was isolated by a functional genomics approach in which we employed 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 DMT1 and co-localized with the chardonnay gene. We identified a nonsense mutation in the chardonnay allele and demonstrated that, while wild-type zebrafish DMT1 protein can transport iron, the truncated protein expressed in chardonnay 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. CiteULike    Connotea    Del.icio.us    Digg    Reddit    Technorati    What's this? This article has been cited by other articles: D. L. Stachura, J. R. Reyes, P. Bartunek, B. H. Paw, L. I. Zon, and D. Traver Zebrafish kidney stromal cell lines support multilineage hematopoiesis Blood, July 9, 2009; 114(2): 279 - 289. [Abstract] [Full Text] [PDF] S. Soe-Lin, S. S. Apte, B. Andriopoulos Jr., M. C. Andrews, M. Schranzhofer, T. Kahawita, D. Garcia-Santos, and P. 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