Literature DB >> 18210110

[Hereditary hemochromatosis, alpha-1-antitrypsin deficiency and Wilson's disease. Pathogenesis, clinical findings and pathways to diagnosis].

H Zhou1, H-P Fischer.   

Abstract

Primary hemochromatosis, alpha-1-antitrypsin (AAT) deficiency, and Wilson's disease are the most common hereditary causes of unclear hepatopathy. Classical primary hemochromatosis (type I) on the basis of a homozygous mutation of the HFE gene, usually presents in adults with increasing hepatocellular siderosis and chronic progressive necroinflammatory liver disease. Homozygous AAT deficiency type PiZZ becomes manifest in newborns as a giant cell hepatitis or findings similar to bile duct atresia, in adults as chronic hepatitis or "cryptogenic cirrhosis". The heterozygous PiZ mutation can lead to PAS-positive hepatocellular AAT deposits increasing over the life time. Immunohistochemical detection of AAT deposits by specific PiZ antibodies is a highly sensitive and specific supplementary method. Molecular analysis of AAT and HFE genes in paraffin-embedded tissue or blood can confirm the diagnosis and allows the zygosity status to be defined. Wilson's disease has to be considered in children and young adults with unexplained histologic findings of chronic hepatitis or steatohepatitis. Rhodanin staining is the most effective histochemical method to detect free copper deposits, but negative staining results do not exclude Wilson's disease. In cases suspected of Wilson's disease further clinical exploration must be initiated. The diagnosis is based on a combination of clinical and biochemical findings, which can be supplemented by mutation analysis of the ATP7B gene.

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Year:  2008        PMID: 18210110     DOI: 10.1007/s00292-007-0954-2

Source DB:  PubMed          Journal:  Pathologe        ISSN: 0172-8113            Impact factor:   1.011


  40 in total

1.  [Long-term substitution in homozygous alpha 1-antitrypsin deficiency. Effect of the proteinase-antiproteinase equilibrium in plasma and sputum].

Authors:  J Braun; S Welle; J van Wees; R Winterhoff; W G Wood; K Dalhoff; K J Wiessmann
Journal:  Dtsch Med Wochenschr       Date:  1990-06-08       Impact factor: 0.628

2.  Chronic liver disease in heterozygous alpha1-antitrypsin deficiency PiZ.

Authors:  H P Fischer; M E Ortiz-Pallardó; Y Ko; C Esch; H Zhou
Journal:  J Hepatol       Date:  2000-12       Impact factor: 25.083

3.  [Liver changes in heterozygote alpha 1-antitrypsin deficiency PiZ].

Authors:  H Zhou; H P Fischer
Journal:  Pathologe       Date:  2000-11       Impact factor: 1.011

Review 4.  Wilson's Disease.

Authors:  Peter Ferenci
Journal:  Clin Gastroenterol Hepatol       Date:  2005-08       Impact factor: 11.382

Review 5.  [Iron as comorbid factor in chronic hepatitis C].

Authors:  Andreas Erhardt; Katarzyna Hauck; Dieter Häussinger
Journal:  Med Klin (Munich)       Date:  2003-12-15

6.  Liver transplantation in 100 children: Cambridge and King's College Hospital series.

Authors:  A Salt; G Noble-Jamieson; N D Barnes; A P Mowat; K Rolles; N Jamieson; P Johnston; P Friend; R Y Calne
Journal:  BMJ       Date:  1992-02-15

7.  Risk of cirrhosis and primary liver cancer in alpha 1-antitrypsin deficiency.

Authors:  S Eriksson; J Carlson; R Velez
Journal:  N Engl J Med       Date:  1986-03-20       Impact factor: 91.245

Review 8.  Alpha-1-antitrypsin deficiency.

Authors:  D H Perlmutter
Journal:  Semin Liver Dis       Date:  1998       Impact factor: 6.115

Review 9.  Alpha 1-antitrypsin deficiency and liver disease: clinical presentation, diagnosis and treatment.

Authors:  M Hussain; G Mieli-Vergani; A P Mowat
Journal:  J Inherit Metab Dis       Date:  1991       Impact factor: 4.982

10.  The liver in adolescents with alpha 1-antitrypsin deficiency.

Authors:  T Sveger; S Eriksson
Journal:  Hepatology       Date:  1995-08       Impact factor: 17.425

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  2 in total

Review 1.  [Lysosomal acid lipase deficiency (LAL-D) : Diagnostic and therapeutic options in an underdiagnosed disease].

Authors:  S Synoracki; S Kathemann; K W Schmid; H Jastrow; H A Baba
Journal:  Pathologe       Date:  2018-05       Impact factor: 1.011

Review 2.  Cryptogenic chronic hepatitis and its changing guise in adults.

Authors:  Albert J Czaja
Journal:  Dig Dis Sci       Date:  2011-06-07       Impact factor: 3.199

  2 in total

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