Literature DB >> 3753845

Severe liver fibrosis in argininosuccinic aciduria.

A Zimmermann, C Bachmann, R Baumgartner.   

Abstract

Hepatomegaly is an important clinical finding in patients with argininosuccinic aciduria (a hereditary defect of the urea cycle enzyme, argininosuccinate lyase [argininosuccinase]). A severe degree of liver fibrosis, almost corresponding to cirrhosis, was observed in liver biopsy material obtained from a boy with this disorder. This observation is of interest in light of the fact that liver fibrosis or cirrhosis are hallmarks of many inheritable phenotypes, and especially of inborn errors of metabolism. Variable degrees of liver fibrosis are noted in other inborn defects of the urea cycle, eg, in ornithine transcarbamylase and carbamoylphosphate synthetase deficiencies. These findings appear to indicate that inheritable defects of urea synthesis may form a group of metabolic disorders prone to cause hepatic fibrosis, or even cirrhosis, as shown in our patient.

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Year:  1986        PMID: 3753845

Source DB:  PubMed          Journal:  Arch Pathol Lab Med        ISSN: 0003-9985            Impact factor:   5.534


  18 in total

1.  Differential expression of the two human arginase genes in hyperargininemia. Enzymatic, pathologic, and molecular analysis.

Authors:  W W Grody; C Argyle; R M Kern; G J Dizikes; E B Spector; A D Strickland; D Klein; S D Cederbaum
Journal:  J Clin Invest       Date:  1989-02       Impact factor: 14.808

2.  Bacterial expression of mutant argininosuccinate lyase reveals imperfect correlation of in-vitro enzyme activity with clinical phenotype in argininosuccinic aciduria.

Authors:  Katharina Engel; Jean-Marc Vuissoz; Sandra Eggimann; Murielle Groux; Christoph Berning; Liyan Hu; Vera Klaus; Dorothea Moeslinger; Saadet Mercimek-Mahmutoglu; Sylvia Stöckler; Bendicht Wermuth; Johannes Häberle; Jean-Marc Nuoffer
Journal:  J Inherit Metab Dis       Date:  2011-06-11       Impact factor: 4.982

3.  Chronic liver disease and impaired hepatic glycogen metabolism in argininosuccinate lyase deficiency.

Authors:  Lindsay C Burrage; Simran Madan; Xiaohui Li; Saima Ali; Mahmoud Mohammad; Bridget M Stroup; Ming-Ming Jiang; Racel Cela; Terry Bertin; Zixue Jin; Jian Dai; Danielle Guffey; Milton Finegold; Sandesh Nagamani; Charles G Minard; Juan Marini; Prakash Masand; Deborah Schady; Benjamin L Shneider; Daniel H Leung; Deeksha Bali; Brendan Lee
Journal:  JCI Insight       Date:  2020-02-27

4.  Requirement of argininosuccinate lyase for systemic nitric oxide production.

Authors:  Ayelet Erez; Sandesh C S Nagamani; Oleg A Shchelochkov; Muralidhar H Premkumar; Philippe M Campeau; Yuqing Chen; Harsha K Garg; Li Li; Asad Mian; Terry K Bertin; Jennifer O Black; Heng Zeng; Yaoping Tang; Anilkumar K Reddy; Marshall Summar; William E O'Brien; David G Harrison; William E Mitch; Juan C Marini; Judy L Aschner; Nathan S Bryan; Brendan Lee
Journal:  Nat Med       Date:  2011-11-13       Impact factor: 53.440

5.  Mitochondrial abnormalities of liver in two children with citrullinaemia.

Authors:  S A Zamora; A Pinto; R B Scott; H G Parsons
Journal:  J Inherit Metab Dis       Date:  1997-08       Impact factor: 4.982

6.  Unstable argininosuccinate lyase in variant forms of the urea cycle disorder argininosuccinic aciduria.

Authors:  Liyan Hu; Amit V Pandey; Cécile Balmer; Sandra Eggimann; Véronique Rüfenacht; Jean-Marc Nuoffer; Johannes Häberle
Journal:  J Inherit Metab Dis       Date:  2015-03-17       Impact factor: 4.982

Review 7.  Optimizing therapy for argininosuccinic aciduria.

Authors:  Sandesh C S Nagamani; Brendan Lee; Ayelet Erez
Journal:  Mol Genet Metab       Date:  2012-07-20       Impact factor: 4.797

8.  A randomized controlled trial to evaluate the effects of high-dose versus low-dose of arginine therapy on hepatic function tests in argininosuccinic aciduria.

Authors:  Sandesh C S Nagamani; Oleg A Shchelochkov; Mary A Mullins; Susan Carter; Brendan C Lanpher; Qin Sun; Soledad Kleppe; Ayelet Erez; E O'Brian Smith; Juan C Marini; Brendan Lee
Journal:  Mol Genet Metab       Date:  2012-09-17       Impact factor: 4.797

9.  AAV gene therapy corrects OTC deficiency and prevents liver fibrosis in aged OTC-knock out heterozygous mice.

Authors:  Lili Wang; Peter Bell; Hiroki Morizono; Zhenning He; Elena Pumbo; Hongwei Yu; John White; Mark L Batshaw; James M Wilson
Journal:  Mol Genet Metab       Date:  2017-03-02       Impact factor: 4.797

10.  Argininosuccinate lyase deficiency: longterm outcome of 13 patients detected by newborn screening.

Authors:  C Ficicioglu; R Mandell; V E Shih
Journal:  Mol Genet Metab       Date:  2009-06-25       Impact factor: 4.797

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