Literature DB >> 15727899

Autosomal dominant hereditary hemochromatosis associated with a novel ferroportin mutation and unique clinical features.

Ronald L Sham1, Pradyumna D Phatak, Carol West, Pauline Lee, Caroline Andrews, Ernest Beutler.   

Abstract

Hereditary hemochromatosis is a common disorder of iron metabolism most frequently associated with mutations in the HFE gene. Hereditary hemochromatosis may be caused by other less common genetic mutations including those in the ferroportin gene. Whereas hereditary hemochromatosis associated with HFE mutations is an autosomal recessive disorder, essentially all cases of hereditary hemochromatosis associated with ferroportin mutations follow an autosomal dominant pattern of inheritance, and most cases are notable for the lack of an elevated transferrin saturation and presence of iron deposition in Kupffer cells. This report describes the clinical and laboratory features of a family with hereditary hemochromatosis associated with a previously unrecognized ferroportin mutation (Cys326Ser). Three generations of the family are described. The disease in this family is notable for young age at onset, elevated transferrin saturation values, and hepatocyte iron deposition. The distinct molecular and clinical features reflect the heterogeneous nature of this disease.

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Year:  2005        PMID: 15727899     DOI: 10.1016/j.bcmd.2004.12.002

Source DB:  PubMed          Journal:  Blood Cells Mol Dis        ISSN: 1079-9796            Impact factor:   3.039


  33 in total

Review 1.  Hepcidin and iron homeostasis.

Authors:  Tomas Ganz; Elizabeta Nemeth
Journal:  Biochim Biophys Acta       Date:  2012-01-26

Review 2.  Modulation of hepcidin to treat iron deregulation: potential clinical applications.

Authors:  Nicole L Blanchette; David H Manz; Frank M Torti; Suzy V Torti
Journal:  Expert Rev Hematol       Date:  2015-12-15       Impact factor: 2.929

Review 3.  Iron overload due to mutations in ferroportin.

Authors:  Ivana De Domenico; Diane McVey Ward; Giovanni Musci; Jerry Kaplan
Journal:  Haematologica       Date:  2006-01       Impact factor: 9.941

4.  Hereditary hemochromatosis due to resistance to hepcidin: high hepcidin concentrations in a family with C326S ferroportin mutation.

Authors:  Ronald L Sham; Pradyumna D Phatak; Elizabeta Nemeth; Tomas Ganz
Journal:  Blood       Date:  2009-07-09       Impact factor: 22.113

5.  Structure-function analysis of ferroportin defines the binding site and an alternative mechanism of action of hepcidin.

Authors:  Sharraya Aschemeyer; Bo Qiao; Deborah Stefanova; Erika V Valore; Albert C Sek; T Alex Ruwe; Kyle R Vieth; Grace Jung; Carla Casu; Stefano Rivella; Mika Jormakka; Bryan Mackenzie; Tomas Ganz; Elizabeta Nemeth
Journal:  Blood       Date:  2017-12-13       Impact factor: 22.113

6.  The molecular basis of ferroportin-linked hemochromatosis.

Authors:  Ivana De Domenico; Diane McVey Ward; Elizabeta Nemeth; Michael B Vaughn; Giovanni Musci; Tomas Ganz; Jerry Kaplan
Journal:  Proc Natl Acad Sci U S A       Date:  2005-06-13       Impact factor: 11.205

Review 7.  The pathophysiology and pharmacology of hepcidin.

Authors:  Piotr Ruchala; Elizabeta Nemeth
Journal:  Trends Pharmacol Sci       Date:  2014-02-17       Impact factor: 14.819

8.  Reduced sensitivity of the ferroportin Q248H mutant to physiological concentrations of hepcidin.

Authors:  Sergei Nekhai; Min Xu; Altreisha Foster; Ishmael Kasvosve; Sharmin Diaz; Roberto F Machado; Oswaldo L Castro; Gregory J Kato; James G Taylor; Victor R Gordeuk
Journal:  Haematologica       Date:  2012-10-12       Impact factor: 9.941

9.  The molecular basis of hepcidin-resistant hereditary hemochromatosis.

Authors:  Augustine Fernandes; Gloria C Preza; Yen Phung; Ivana De Domenico; Jerry Kaplan; Tomas Ganz; Elizabeta Nemeth
Journal:  Blood       Date:  2009-04-21       Impact factor: 22.113

10.  Non-HFE hemochromatosis.

Authors:  Paulo Caleb Júnior de Lima Santos; Carla Luana Dinardo; Rodolfo Delfini Cançado; Isolmar Tadeu Schettert; José Eduardo Krieger; Alexandre Costa Pereira
Journal:  Rev Bras Hematol Hemoter       Date:  2012
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