| Literature DB >> 16439678 |
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.Entities:
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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