Literature DB >> 16819392

Variable degree of liver involvement in siblings with PiZZ alpha-1-antitrypsin deficiency-related liver disease.

R Hinds1, A Hadchouel, N P Shanmugham, A Al-Hussaini, S Chambers, P Cheeseman, G Mieli-Vergani, N Hadzić.   

Abstract

PiZZ alpha-1-antitrypsin deficiency is the commonest genetic cause of chronic liver disease, but only 10-15% of PiZZ individuals develop liver disease in childhood. Studies have demonstrated varying patterns of disease progression within siblings with the PiZZ phenotype. We retrospectively analysed the case-notes of all patients diagnosed with PiZZ A1ATD between 1978-2002 and compared the pattern of liver disease between affected siblings. We identified 29 families with more than 1 child with the PiZZ phenotype. Twenty-one (72%) PiZZ siblings of the 29 probands had liver disease, which was concordant for severity in 6 (29%), while 8 (28%) had no liver involvement. Five of 7 children requiring liver transplantation had siblings with no persistent liver dysfunction. This study suggests that there is a variable degree of liver involvement in siblings with PiZZ A1ATD-related liver disease and environmental and/or other genetic factors must be involved in determining disease severity.

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Year:  2006        PMID: 16819392     DOI: 10.1097/01.mpg.0000226370.09085.39

Source DB:  PubMed          Journal:  J Pediatr Gastroenterol Nutr        ISSN: 0277-2116            Impact factor:   2.839


  5 in total

Review 1.  Diagnosis and management of patients with α1-antitrypsin (A1AT) deficiency.

Authors:  David R Nelson; Jeffrey Teckman; Adrian M Di Bisceglie; David A Brenner
Journal:  Clin Gastroenterol Hepatol       Date:  2011-12-23       Impact factor: 11.382

2.  Prevalence of genetic polymorphisms in the promoter region of the alpha-1 antitrypsin (SERPINA1) gene in chronic liver disease: a case control study.

Authors:  Karin F Kok; René H te Morsche; Martijn G H van Oijen; Joost P H Drenth
Journal:  BMC Gastroenterol       Date:  2010-02-20       Impact factor: 3.067

Review 3.  Alpha-1 antitrypsin deficiency: a conformational disease associated with lung and liver manifestations.

Authors:  C M Greene; S D W Miller; T Carroll; C McLean; M O'Mahony; M W Lawless; S J O'Neill; C C Taggart; N G McElvaney
Journal:  J Inherit Metab Dis       Date:  2008-01-16       Impact factor: 4.982

4.  Genotyping diagnosis of alpha-1 antitrypsin deficiency in Saudi adults with liver cirrhosis.

Authors:  Noura Al-Jameil; Amina A Hassan; Ahlam Buhairan; Rana Hassanato; Sree R Isac; Maram Al-Otaiby; Basmah Al-Maarik; Iman Al-Ajeyan
Journal:  Medicine (Baltimore)       Date:  2017-02       Impact factor: 1.889

5.  Hepatic fibrinogen storage disease due to the fibrinogen γ375 Arg → Trp mutation "fibrinogen Aguadilla" is present in Arabs.

Authors:  Abdulrahman Al-Hussaini; Abdulhadi Altalhi; Imad El Hag; Hussa AlHussaini; Paola Francalanci; Isabella Giovannoni; Francesco Callea
Journal:  Saudi J Gastroenterol       Date:  2014 Jul-Aug       Impact factor: 2.485

  5 in total

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