Literature DB >> 26946192

Is severe progressive liver disease caused by alpha-1-antitrypsin deficiency more common in children or adults?

Andrew S Chu1,2, Kapil B Chopra3, David H Perlmutter1,4,2.   

Abstract

The classical form of alpha-1-antitrypsin deficiency (A1ATD) is known to cause liver disease in children and adults, but there is relatively little information about the risk of severe, progressive liver disease and the need for liver transplantation. To better understand how newly evolving pharmacological, genetic, and cellular therapies may be targeted according to risk for progressive liver disease, we sought to determine the age distribution of A1ATD as a cause of severe liver disease, as defined by the need for liver transplantation. Using 3 US liver transplantation databases for the period 1991-2012, we found 77.2% of 1677 liver transplants with a reported diagnosis of A1ATD were adults. The peak age range was 50-64 years. Using 2 of the databases which included specific A1AT phenotypes, we found that many of these adults who undergo liver transplantation with A1ATD as the diagnosis are heterozygotes and have other potential causes of liver disease, most notably obesity and ethanol abuse. However, even when these cases are excluded and only ZZ and SZ phenotypes are considered, severe liver disease requiring transplantation is more than 2.5 times as likely in adults. The analysis also showed a markedly increased risk for males. In the pediatric group, almost all of the transplants are done in children less than 5 years of age. In conclusion, A1ATD causes progressive liver disease most commonly in adults with males in the highest risk category. In the pediatric group, children less than 5 years of age are highest in risk. These results suggest that A1ATD most commonly causes liver disease by mechanisms similar to age-dependent degenerative diseases and more rarely in children by powerful modifiers. Liver Transplantation 22 886-894 2016 AASLD.
© 2016 American Association for the Study of Liver Diseases.

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Year:  2016        PMID: 26946192     DOI: 10.1002/lt.24434

Source DB:  PubMed          Journal:  Liver Transpl        ISSN: 1527-6465            Impact factor:   5.799


  13 in total

Review 1.  Alpha-1 Antitrypsin Deficiency-Mediated Liver Toxicity: Why Do Some Patients Do Poorly? What Do We Know So Far?

Authors:  Marion Bouchecareilh
Journal:  Chronic Obstr Pulm Dis       Date:  2020-07

Review 2.  Update on Alpha-1 Antitrypsin Deficiency in Liver Disease.

Authors:  Praveena Narayanan; Pramod K Mistry
Journal:  Clin Liver Dis (Hoboken)       Date:  2020-06-30

3.  Expression of mitochondrial membrane-linked SAB determines severity of sex-dependent acute liver injury.

Authors:  Sanda Win; Robert Wm Min; Christopher Q Chen; Jun Zhang; Yibu Chen; Meng Li; Ayako Suzuki; Manal F Abdelmalek; Ying Wang; Mariam Aghajan; Filbert Wm Aung; Anna Mae Diehl; Roger J Davis; Tin A Than; Neil Kaplowitz
Journal:  J Clin Invest       Date:  2019-12-02       Impact factor: 14.808

4.  Non-invasive assessment for alpha-1 antitrypsin deficiency-associated liver disease: new insights on steatosis and fibrosis in Pi*ZZ carriers.

Authors:  Siyer Roohani; Frank Tacke
Journal:  Transl Gastroenterol Hepatol       Date:  2019-12-12

5.  CHOP and c-JUN up-regulate the mutant Z α1-antitrypsin, exacerbating its aggregation and liver proteotoxicity.

Authors:  Sergio Attanasio; Rosa Ferriero; Gwladys Gernoux; Rossella De Cegli; Annamaria Carissimo; Edoardo Nusco; Severo Campione; Jeffrey Teckman; Christian Mueller; Pasquale Piccolo; Nicola Brunetti-Pierri
Journal:  J Biol Chem       Date:  2020-07-28       Impact factor: 5.157

6.  Development of an RNAi therapeutic for alpha-1-antitrypsin liver disease.

Authors:  Christine I Wooddell; Keith Blomenkamp; Ryan M Peterson; Vladimir M Subbotin; Christian Schwabe; James Hamilton; Qili Chu; Dawn R Christianson; Julia O Hegge; John Kolbe; Holly L Hamilton; Maria F Branca-Afrazi; Bruce D Given; David L Lewis; Edward Gane; Steven B Kanner; Jeffrey H Teckman
Journal:  JCI Insight       Date:  2020-06-18

7.  α1-antitrypsin Deficiency: A Misfolded Secretory Protein Variant with Unique Effects on the Endoplasmic Reticulum.

Authors:  David H Perlmutter
Journal:  Endoplasmic Reticulum Stress Dis       Date:  2016-09-19

8.  Heterozygosity for the alpha-1-antitrypsin Z allele in cirrhosis is associated with more advanced disease.

Authors:  Benedikt Schaefer; Mattias Mandorfer; André Viveiros; Armin Finkenstedt; Peter Ferenci; Stefan Schneeberger; Herbert Tilg; Heinz Zoller
Journal:  Liver Transpl       Date:  2018-05-14       Impact factor: 5.799

9.  Hepatocyte proteomes reveal the role of protein disulfide isomerase 4 in alpha 1-antitrypsin deficiency.

Authors:  Esra Karatas; Anne-Aurélie Raymond; Céline Leon; Jean-William Dupuy; Sylvaine Di-Tommaso; Nathalie Senant; Sophie Collardeau-Frachon; Mathias Ruiz; Alain Lachaux; Frédéric Saltel; Marion Bouchecareilh
Journal:  JHEP Rep       Date:  2021-04-24

Review 10.  Liver Disease in Alpha-1 Antitrypsin Deficiency: Current Approaches and Future Directions.

Authors:  Ellen L Mitchell; Zahida Khan
Journal:  Curr Pathobiol Rep       Date:  2017-07-10
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