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Blood, 19 February 2009, Vol. 113, No. 8, pp. 1805-1808. Prepublished online as a Blood First Edition Paper on October 27, 2008; DOI 10.1182/blood-2007-11-120402. This Article Full Text (PDF) Supplementary Tables All Versions of this Article: blood-2007-11-120402v1 113/8/1805    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 CrossRef Citing Articles via Google Scholar Google Scholar Articles by Lambe, T. Articles by Cornall, R. J Search for Related Content PubMed PubMed Citation Articles by Lambe, T. Articles by Cornall, R. J Social Bookmarking                   What's this?  Previous Article  |  Next Article  Submitted November 8, 2007 Accepted July 24, 2008 Identification of a Steap3 endosomal targeting motif essential for normal iron metabolism Teresa Lambe, Robert J Simpson, Sara Dawson, Tiphaine Bouriez-Jones, Tanya L. Crockford, Michelle Lepherd, Gladys O. Latunde-Dada, Hannah Robinson, Kishor B. Raja, Dean R. Campagna, Guadalupe Villarreal Jr, J. Clive Ellory, Christopher C Goodnow, Mark D. Fleming, Andrew T McKie, and Richard J Cornall* Henry Wellcome Building for Molecular Physiology, Oxford University, Oxford, United Kingdom Department of Biochemistry and Nutrition Sciences Research Division, King's College London, London, United Kingdom Australian Cancer Research Foundation Genetics Laboratory, John Curtin School of Medical Research, Australian National University, Canberra, Australia Department of Physiology, Anatomy and Genetics, Oxford University, Oxford, United Kingdom Department of Clinical Biochemistry, King's College London, London, United Kingdom Department of Pathology, Children's Hospital and Harvard Medical School, Boston, MA, United States * Corresponding author; email: rcornall{at}ccmp.ox.ac.uk. Hereditary forms of iron-deficiency anemia, including animal models, have taught us much about the normal physiological control of iron metabolism. However, the discovery of new informative mutants is limited by the natural mutation frequency. To address this limitation, we have developed a screen for heritable abnormalities of red blood cell morphology in mice with single nucleotide changes induced by the chemical mutagen ethylnitrosourea (ENU). We now describe the first strain, fragile-red, with hypochromic microcytic anemia due to a Y228H substitution in the ferrireductase Steap3 (Steap3Y288H). Analysis of the Steap3Y288H mutant identifies a conserved motif required for targeting Steap3 to internal compartments, and highlights how phenotypic screens linked to mutagenesis can identify new functional variants in erythropoiesis and ascribe function to previously unidentified motifs. CiteULike    Connotea    Del.icio.us    Digg    Reddit    Technorati    What's this?

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