Literature DB >> 22093335

A novel Y231del mutation of HFE in hereditary haemochromatosis provides in vivo evidence that the Huh-7 is a human haemochromatotic cell line.

Atsuko Takano1, Hideki Niimi, Yoshinari Atarashi, Takuro Sawasaki, Teiichi Terasaki, Tomoyuki Nakabayashi, Isao Kitajima, Kazuyuki Tobe, Terumi Takahara.   

Abstract

Hereditary haemochromatosis (HH), which is mainly associated with a C282Y polymorphism in HFE, is common among Caucasians of north European descent, but is very rare among Asians. Herein, we report a 43-year-old Japanese man who was diagnosed as having HH. A laboratory examination revealed an elevated serum iron level (280 μg/dl), hyperferritinemia (1698 ng/ml) and a low serum level of hepcidin-25 (4.0 ng/ml). Abdominal magnetic resonance imaging revealed findings suggestive of iron accumulation in the liver and pancreas. HFE gene sequencing in the patient revealed a novel homozygous TAC nucleotide deletion (c. 691_693del) responsible for the loss of a tyrosine at position 231 (p. Y231del) of the HFE protein. This homozygous Y231del mutation was recently found in the Huh-7 hepatoma cell line and was shown to prevent the translocation of HFE to the cell surface. This clinical case provides in vivo evidence suggesting that Huh-7 is undoubtedly a human haemochromatotic cell line and, as such, is a valuable tool for investigating the pathogenesis of HFE-related HH in humans.
© 2011 John Wiley & Sons A/S.

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Year:  2011        PMID: 22093335     DOI: 10.1111/j.1478-3231.2011.02620.x

Source DB:  PubMed          Journal:  Liver Int        ISSN: 1478-3223            Impact factor:   5.828


  4 in total

Review 1.  The mechanisms of systemic iron homeostasis and etiology, diagnosis, and treatment of hereditary hemochromatosis.

Authors:  Hiroshi Kawabata
Journal:  Int J Hematol       Date:  2017-11-13       Impact factor: 2.490

2.  Iranian hereditary hemochromatosis patients: baseline characteristics, laboratory data and gene mutations.

Authors:  Farhad Zamani; Zohreh Bagheri; Maryam Bayat; Seyed-Mohammad Fereshtehnejad; Ali Basi; Hossein Najmabadi; Hossein Ajdarkosh
Journal:  Med Sci Monit       Date:  2012-10

Review 3.  Pathophysiological consequences and benefits of HFE mutations: 20 years of research.

Authors:  Ina Hollerer; André Bachmann; Martina U Muckenthaler
Journal:  Haematologica       Date:  2017-03-09       Impact factor: 9.941

4.  A 10-year Follow-up Study of a Japanese Family with Ferroportin Disease A: Mild Iron Overload with Mild Hyperferritinemia Co-occurring with Hyperhepcidinemia May Be Benign.

Authors:  Hisao Hayashi; Motoyoshi Yano; Naohito Urawa; Akane Mizutani; Shima Hamaoka; Jun Araki; Yuji Kojima; Yutaka Naito; Ayako Kato; Yasuaki Tatsumi; Koichi Kato
Journal:  Intern Med       Date:  2018-05-18       Impact factor: 1.271

  4 in total

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