Necrotic foci, elevated chemokines and infiltrating neutrophils in the liver of glycogen storage disease type Ia

Journal of Hepatology
So Youn KimJanice Y Chou

Abstract

Glycogen storage disease type Ia (GSD-Ia) patients manifest the long-term complication of hepatocellular adenoma (HCA) of unknown etiology. We showed previously that GSD-Ia mice exhibit neutrophilia and elevated serum cytokine levels. This study was conducted to evaluate whether human GSD-Ia patients exhibit analogous increases and whether in GSD-Ia mice a correlation exists between immune abnormalities and, biochemical and histological alterations in the liver. Differential leukocyte counts and cytokine levels were investigated in GSD-Ia patients. Hepatic chemokine production, neutrophil infiltration, and histological abnormalities were investigated in GSD-Ia mice. We show that GSD-Ia patients exhibit increased peripheral neutrophil counts and serum interleukin-8 (IL-8). Compared to normal subjects, HCA-bearing GSD-Ia patients have a 2.8-fold higher serum IL-8 concentration, while GSD-Ia patients without HCA have a 1.4-fold higher concentration. Hepatic injury in GSD-Ia mice is evidenced by necrotic foci, markedly elevated infiltrating neutrophils, and increased hepatic production of chemokines. Peripheral neutrophilia and elevated serum chemokines are characteristic of GSD-Ia with HCA-bearing GSD-Ia patients having the highes...Continue Reading

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Citations

Sep 17, 2009·Molecules and Cells·So Youn Kim, Yun Soo Bae
Jun 24, 2010·Molecular Therapy : the Journal of the American Society of Gene Therapy·Albert GrinshpunEithan Galun
Apr 15, 2010·Molecular Therapy : the Journal of the American Society of Gene Therapy·Wai Han YiuJanice Y Chou
Feb 15, 2011·Journal of Biomedicine & Biotechnology·Andrew SpechtThomas J Conlon
Oct 31, 2014·Genetics in Medicine : Official Journal of the American College of Medical Genetics·Priya S KishnaniMichael S Watson
Apr 21, 2011·Expert Opinion on Biological Therapy·Janice Y Chou, Brian C Mansfield
Jun 23, 2009·Trends in Endocrinology and Metabolism : TEM·D D KoeberlY-T Chen
Mar 17, 2012·Hepatology : Official Journal of the American Association for the Study of Liver Diseases·Young Mok LeeJanice Y Chou
Dec 26, 2012·Biochimica Et Biophysica Acta·Paola MarcolongoAngelo Benedetti
Oct 27, 2010·Nature Reviews. Endocrinology·Janice Y ChouBrian C Mansfield
Aug 26, 2014·Disease Models & Mechanisms·Roberta ResazAlessandra Eva
May 1, 2021·International Journal of Molecular Sciences·Alberto Molares-VilaCarmen Rodríguez-Cerdeira

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