| Literature DB >> 16819392 |
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.Entities:
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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