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Siao, K.

Publications and source records attributed to Siao, K..

2 recordsLinked to original sources

iPSC-Derived Hepatocytes from Patients with MASLD Exhibit Early Mitochondrial Dysfunction

Background & AimsA hallmark of metabolic dysfunction-associated steatotic liver disease (MASLD) is a decline in the ability of hepatocyte mitochondria to adapt to excess lipid. This leads to the production of reactive oxygen species (ROS) and the instigation of a vicious cycle of further mitochondrial damage and cellular dysfunction that promotes disease progression. In this study, we investigated whether induced pluripotent stem cells (iPSCs) from MASLD patients exhibit features of mitochondrial dysfunction when differentiated to hepatocyte-like cells (iPSC-Heps). MethodsiPSCs from 10 MASLD patients and 10 healthy control subjects genotyped for the I148M variant of PNPLA3 were differentiated to iPSC-Heps. Mitochondrial mass and function were assessed under basal culture conditions and following short-term exposure to exogenous palmitate. Outcomes included gene expression, mitochondrial oxygen consumption, ROS production and cellular energy status. ResultsiPSC-Heps from MASLD patients spontaneously accrued more lipid than control iPSC-Heps. Mitochondrial content was similar in MASLD and control iPSC-Heps, but MASLD iPSC-Heps displayed significant differences in mitochondrial function including a decrease in oxygen consumption rate when challenged with palmitate. Antioxidant gene expression was increased at baseline in MASLD vs. control iPSC-Heps, and MASLD iPSC-Heps produced more ROS and less ATP than controls after palmitate treatment. Differences persisted even when controlling for PNPLA3 genotype. ConclusionsiPSC-Heps from MASLD patients exhibit mitochondrial alterations characteristic of their diseased origin. The degree of mitochondrial dysfunction seen in MASLD iPSC-Heps is reminiscent of that described clinically in early MASLD, prior to progression to steatohepatitis. Mitochondrial alterations in MASLD iPSC-Heps occur independently of PNPLA3 genotype.

cell biology↗

Hepatocyte-specific deletion of XBP1 sensitizes mice to liver injury through hyperactivation of IRE1α

X-box binding protein-1 (XBP1) is a transcription factor that plays a central role in controlling cellular responses to endoplasmic reticulum (ER) stress. Under stress conditions, the transcriptionally active form of XBP1 is generated by unique splicing of Xbp1 mRNA by the ER-resident protein inositol-requiring enzyme-1 (IRE1). Genetic deletion of XBP1 has multiple consequences: some resulting from the loss of the transcription factor per se, and others related to compensatory upstream activation of IRE1. The objective of the current study was to investigate the effects of XBP1 deletion in adult mouse liver and determine to what extent they are direct or indirect. XBP1 was deleted specifically from hepatocytes in adult Xbp1fl/fl mice using AAV8-Transthyretin-Cre (Xbp1{Delta}hep). Xbp1{Delta}hep mice exhibited no liver disease at baseline, but developed acute biochemical and histologic liver injury in response to a dietary challenge with fructose for 4 wk. Fructose-mediated liver injury in Xbp1{Delta}hep mice coincided with heightened IRE1 activity, as demonstrated by cJun phosphorylation and regulated IRE1 -dependent RNA decay (RIDD). Activation of eIF2 was also evident, with associated up-regulation of the pro-apoptotic molecules CHOP, BIM and PUMA. To determine whether the adverse consequences of liver-specific XBP1 deletion were due to XBP1 loss or heightened IRE1 activity, we repeated a fructose challenge in mice with liver-specific deletion of both XBP1 and IRE1 (Xbp1{Delta}hep;IRE1{Delta}hep). Xbp1{Delta}hep;IRE1{Delta}hep mice were protected from fructose-mediated liver injury and failed to exhibit any of the signs of ER stress seen in mice lacking XBP1 alone. The protective effect of IRE1 deletion persisted even with long-term exposure to fructose. Xbp1{Delta}hep mice developed liver fibrosis at 16 wk, but Xbp1{Delta}hep;IRE1{Delta}hep mice did not. Overall, the results indicate that the deleterious effects of hepatocyte-specific XBP1 deletion are due primarily to hyperactivation of IRE1. They support further exploration of IRE1 as a contributor to acute and chronic liver diseases.

cell biology↗