Literature DB >> 10673304

Incidence of liver disease in people with HFE mutations.

G Willis1, J Z Wimperis, R Lonsdale, I W Fellows, M A Watson, L M Skipper, B A Jennings.   

Abstract

BACKGROUND: Most patients with haemochromatosis have mutations of the HFE gene. However, the risk to people with HFE mutations of developing disease manifestations of haemochromatosis is not known. AIMS: To determine the risk of developing cirrhosis and liver cancer in individuals with HFE mutations in a population where few people were being treated for haemochromatosis.
METHODS: 215 archive biopsy specimens of liver cancer (n=34) and cirrhosis (n=190) were retrieved from histology archives. Blood samples from 1000 individuals from the normal population were also collected. DNA was extracted from the biopsy specimens and exons 2 and 4 of the HFE gene were amplified using polymerase chain reaction. The products were analysed for the C282Y (845A) and H63D (187G) mutations.
RESULTS: Three (8.8%) patients from the liver cancer group were homozygous for the C282Y mutation. Five (2.6%) patients from the cirrhosis group were homozygous for the C282Y mutation. One case fell in both the liver cancer and cirrhosis groups. C282Y homozygosity was thus significantly more frequent in both groups than in the normal population. These 215 cases are representative of a population of about 250 000 over 20 years. During this period we estimate that about 260 births or deaths of C282Y homozygous individuals occurred within this population.
CONCLUSIONS: A diagnosis of liver cancer or cirrhosis is rare in the lifetime of individuals from this population who are homozygous for the C282Y mutation (2.5%; upper 95% confidence interval (CI) = 8%). Similarly liver disease is rare among C282Y/H63D compound heterozygotes (1%; upper 95% CI = 3.5%).

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Year:  2000        PMID: 10673304      PMCID: PMC1727850          DOI: 10.1136/gut.46.3.401

Source DB:  PubMed          Journal:  Gut        ISSN: 0017-5749            Impact factor:   23.059


  22 in total

1.  Global prevalence of putative haemochromatosis mutations.

Authors:  A T Merryweather-Clarke; J J Pointon; J D Shearman; K J Robson
Journal:  J Med Genet       Date:  1997-04       Impact factor: 6.318

2.  A novel MHC class I-like gene is mutated in patients with hereditary haemochromatosis.

Authors:  J N Feder; A Gnirke; W Thomas; Z Tsuchihashi; D A Ruddy; A Basava; F Dormishian; R Domingo; M C Ellis; A Fullan; L M Hinton; N L Jones; B E Kimmel; G S Kronmal; P Lauer; V K Lee; D B Loeb; F A Mapa; E McClelland; N C Meyer; G A Mintier; N Moeller; T Moore; E Morikang; C E Prass; L Quintana; S M Starnes; R C Schatzman; K J Brunke; D T Drayna; N J Risch; B R Bacon; R K Wolff
Journal:  Nat Genet       Date:  1996-08       Impact factor: 38.330

3.  Analysis of the cost of population screening for haemochromatosis using biochemical and genetic markers.

Authors:  M L Bassett; B A Leggett; J W Halliday; S Webb; L W Powell
Journal:  J Hepatol       Date:  1997-09       Impact factor: 25.083

4.  A high prevalence of HLA-H 845A mutations in hemochromatosis patients and the normal population in eastern England.

Authors:  G Willis; B A Jennings; E Goodman; I W Fellows; J Z Wimperis
Journal:  Blood Cells Mol Dis       Date:  1997-08       Impact factor: 3.039

5.  Haemochromatosis gene mutation in hepatocellular cancer.

Authors:  G Willis; J Z Wimperis; R Lonsdale; B A Jennings
Journal:  Lancet       Date:  1997-08-23       Impact factor: 79.321

6.  Long-term survival analysis in hereditary hemochromatosis.

Authors:  P C Adams; M Speechley; A E Kertesz
Journal:  Gastroenterology       Date:  1991-08       Impact factor: 22.682

7.  Mutation analysis in hereditary hemochromatosis.

Authors:  E Beutler; T Gelbart; C West; P Lee; M Adams; R Blackstone; P Pockros; M Kosty; C P Venditti; P D Phatak; N K Seese; K A Chorney; A E Ten Elshof; G S Gerhard; M Chorney
Journal:  Blood Cells Mol Dis       Date:  1996       Impact factor: 3.039

8.  The incidence of rheumatoid arthritis in the United Kingdom: results from the Norfolk Arthritis Register.

Authors:  D P Symmons; E M Barrett; C R Bankhead; D G Scott; A J Silman
Journal:  Br J Rheumatol       Date:  1994-08

9.  Survival and prognostic factors in 212 Italian patients with genetic hemochromatosis.

Authors:  S Fargion; C Mandelli; A Piperno; B Cesana; A L Fracanzani; M Fraquelli; P A Bianchi; G Fiorelli; D Conte
Journal:  Hepatology       Date:  1992-04       Impact factor: 17.425

10.  Long-term survival in patients with hereditary hemochromatosis.

Authors:  C Niederau; R Fischer; A Pürschel; W Stremmel; D Häussinger; G Strohmeyer
Journal:  Gastroenterology       Date:  1996-04       Impact factor: 22.682

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  11 in total

1.  Deaths attributed to haemochromatosis are rare in Britain.

Authors:  G Willis; I W Fellows; J Z Wimperis
Journal:  BMJ       Date:  2000-04-22

2.  Iron loading and morbidity among relatives of HFE C282Y homozygotes identified either by population genetic testing or presenting as patients.

Authors:  C A McCune; D Ravine; K Carter; H A Jackson; D Hutton; J Hedderich; M Krawczak; M Worwood
Journal:  Gut       Date:  2005-09-20       Impact factor: 23.059

3.  Normal iron metabolism and the pathophysiology of iron overload disorders.

Authors:  Chiang W Siah; John Ombiga; Leon A Adams; Debbie Trinder; John K Olynyk
Journal:  Clin Biochem Rev       Date:  2006-02

4.  Association between C282Y and H63D mutations of the HFE gene with hepatocellular carcinoma in European populations: a meta-analysis.

Authors:  Fei Jin; Li-Shuai Qu; Xi-Zhong Shen
Journal:  J Exp Clin Cancer Res       Date:  2010-03-02

Review 5.  Screening for hemochromatosis: patients with liver disease, families, and populations.

Authors:  Sumedha P Galhenage; Charlie H Viiala; John K Olynyk
Journal:  Curr Gastroenterol Rep       Date:  2004-02

6.  Hepatic iron overload identified by magnetic resonance imaging-based T2* is a predictor of non-diagnostic elastography.

Authors:  Hassan M Ghoz; Paul T Kröner; Fernando F Stancampiano; Andrew W Bowman; Prakash Vishnu; Michael G Heckman; Nancy N Diehl; Ethan McLeod; Naveed Nikpour; William C Palmer
Journal:  Quant Imaging Med Surg       Date:  2019-06

7.  Hepatocellular carcinoma and the penetrance of HFE C282Y mutations: a cross sectional study.

Authors:  Gavin Willis; Vicky Bardsley; Ian W Fellows; Ray Lonsdale; Jennie Z Wimperis; Barbara A Jennings
Journal:  BMC Gastroenterol       Date:  2005-06-01       Impact factor: 3.067

Review 8.  Evidence for the Influence of the Iron Regulatory MHC Class I Molecule HFE on Tumor Progression in Experimental Models and Clinical Populations.

Authors:  Cody Weston; James Connor
Journal:  Transl Oncogenomics       Date:  2014-12-04

9.  Clinical burden of liver disease from hemochromatosis at an academic medical center.

Authors:  Sergio A Sánchez-Luna; Kyle E Brown
Journal:  Hepatol Commun       Date:  2017-05-18

Review 10.  Diabetes in HFE Hemochromatosis.

Authors:  James C Barton; Ronald T Acton
Journal:  J Diabetes Res       Date:  2017-02-26       Impact factor: 4.011

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