Language

English

Publication Date

11-1-2025

Journal

Nature Metabolism

DOI

10.1038/s42255-025-01399-3

PMID

41238906

PMCID

PMC12638245

PubMedCentral® Posted Date

11-14-2025

PubMedCentral® Full Text Version

Post-print

Abstract

Citrin deficiency (CD) is caused by the inactivation of SLC25A13, a mitochondrial membrane protein required to move electrons from cytosolic NADH to the mitochondrial matrix in hepatocytes. People with CD do not like sweets. Here we show that SLC25A13 loss causes the accumulation of glycerol-3-phosphate (G3P), which activates the carbohydrate response element-binding protein (ChREBP) to transcribe FGF21, which acts in the brain to restrain intake of sweets and alcohol and to transcribe key genes driving lipogenesis. Mouse and human data suggest that G3P-ChREBP is a mechanistic component of the Randle Cycle that contributes to metabolic-dysfunction-associated steatotic liver disease and forms part of a system that communicates metabolic states from the liver to the brain in a manner that alters food and alcohol choices. The data provide a framework for understanding FGF21 induction in varied conditions, suggest ways to develop FGF21-inducing drugs and suggest potential drug candidates for lean metabolic-dysfunction-associated steatotic liver disease and support of urea cycle function in CD.

Keywords

Fibroblast Growth Factors, Animals, Lipogenesis, Mice, Humans, Glycerophosphates, Basic Helix-Loop-Helix Leucine Zipper Transcription Factors, Calcium-Binding Proteins, Organic Anion Transporters, Transcription, Genetic, Fatty Liver, Mitochondrial Membrane Transport Proteins, Mice, Knockout, Mice, Inbred C57BL, Metabolic diseases, Gene regulation, Transcriptional regulatory elements, Metabolism, Liver

Published Open-Access

yes

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