Literature DB >> 16439678

Two new human DMT1 gene mutations in a patient with microcytic anemia, low ferritinemia, and liver iron overload.

Carole Beaumont1, Jean Delaunay, Gilles Hetet, Bernard Grandchamp, Mariane de Montalembert, Gil Tchernia.   

Abstract

DMT1 mediates the pH-dependent uptake of Fe(2+) from the diet in duodenal enterocytes and in most other cells. It transfers iron from the endosomes to the cytosol following the uptake of the transferrin-transferrin receptor complex. DMT1 mutations are responsible for severe hypochromic microcytic anemia in rodents and in 2 human patients described recently. We report a compound heterozygote for 2 new DMT1 mutations, associated with microcytic anemia from birth and progressive liver iron overload. The first mutation is a GTG deletion in exon 5, leading to the V114 in-frame deletion in transmembrane domain 2, and the second is a G --> T substitution in exon 8 leading to the G212V replacement in transmembrane domain 5. Together with the 2 previously reported cases, this patient defines a new syndrome of congenital microcytic hypochromic anemia, poorly responsive to oral iron treatment, with liver iron overload associated paradoxically with normal to moderately elevated serum ferritin levels.

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Year:  2006        PMID: 16439678     DOI: 10.1182/blood-2005-10-4269

Source DB:  PubMed          Journal:  Blood        ISSN: 0006-4971            Impact factor:   22.113


  42 in total

1.  Belgrade rats display liver iron loading.

Authors:  Khristy Thompson; Ramon M Molina; Joseph D Brain; Marianne Wessling-Resnick
Journal:  J Nutr       Date:  2006-12       Impact factor: 4.798

2.  DMT1 mutation: response of anemia to darbepoetin administration and implications for iron homeostasis.

Authors:  Dagmar Pospisilova; Martha P Mims; Elizabeta Nemeth; Tomas Ganz; Josef T Prchal
Journal:  Blood       Date:  2006-07-01       Impact factor: 22.113

Review 3.  Forging a field: the golden age of iron biology.

Authors:  Nancy C Andrews
Journal:  Blood       Date:  2008-07-15       Impact factor: 22.113

Review 4.  The long history of iron in the Universe and in health and disease.

Authors:  Alex D Sheftel; Anne B Mason; Prem Ponka
Journal:  Biochim Biophys Acta       Date:  2011-08-09

5.  Crystal structure of a SLC11 (NRAMP) transporter reveals the basis for transition-metal ion transport.

Authors:  Ines A Ehrnstorfer; Eric R Geertsma; Els Pardon; Jan Steyaert; Raimund Dutzler
Journal:  Nat Struct Mol Biol       Date:  2014-10-19       Impact factor: 15.369

Review 6.  Manganese transport in eukaryotes: the role of DMT1.

Authors:  Catherine Au; Alexandre Benedetto; Michael Aschner
Journal:  Neurotoxicology       Date:  2008-05-14       Impact factor: 4.294

7.  Iron loading impairs lipoprotein lipase activity and promotes hypertriglyceridemia.

Authors:  Jonghan Kim; Xuming Jia; Peter D Buckett; Sihao Liu; Chih-Hao Lee; Marianne Wessling-Resnick
Journal:  FASEB J       Date:  2012-12-14       Impact factor: 5.191

Review 8.  Molecular basis of inherited microcytic anemia due to defects in iron acquisition or heme synthesis.

Authors:  Achille Iolascon; Luigia De Falco; Carole Beaumont
Journal:  Haematologica       Date:  2009-01-30       Impact factor: 9.941

9.  Transmembrane topology of the mammalian Slc11a2 iron transporter.

Authors:  Maciej Czachorowski; Steven Lam-Yuk-Tseung; Mathieu Cellier; Philippe Gros
Journal:  Biochemistry       Date:  2009-09-08       Impact factor: 3.162

10.  SMF-1, SMF-2 and SMF-3 DMT1 orthologues regulate and are regulated differentially by manganese levels in C. elegans.

Authors:  Catherine Au; Alexandre Benedetto; Joel Anderson; Arnaud Labrousse; Keith Erikson; Jonathan J Ewbank; Michael Aschner
Journal:  PLoS One       Date:  2009-11-18       Impact factor: 3.240

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