UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation

Summary: Ribosomal UFMylation contributes to ribosome heterogeneity and is associated with ribosome-associated quality control at the endoplasmic reticulum. However, the specific pathophysiological functions of ribosomal UFMylation remain unknown. In this study, we systematically demonstrate the sig...

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Main Authors: Shuchun Yang, Li Wang, Ran Gao, Yanchang Li, Duo Zhang, Chenxi Wang, Guang Liu, Jie Na, Ping Xu, Xiaoyue Wang, Yuyan Jia, Yue Huang
Format: Article
Language:English
Published: Elsevier 2025-05-01
Series:Cell Reports
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Online Access:http://www.sciencedirect.com/science/article/pii/S2211124725004577
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author Shuchun Yang
Li Wang
Ran Gao
Yanchang Li
Duo Zhang
Chenxi Wang
Guang Liu
Jie Na
Ping Xu
Xiaoyue Wang
Yuyan Jia
Yue Huang
author_facet Shuchun Yang
Li Wang
Ran Gao
Yanchang Li
Duo Zhang
Chenxi Wang
Guang Liu
Jie Na
Ping Xu
Xiaoyue Wang
Yuyan Jia
Yue Huang
author_sort Shuchun Yang
collection DOAJ
description Summary: Ribosomal UFMylation contributes to ribosome heterogeneity and is associated with ribosome-associated quality control at the endoplasmic reticulum. However, the specific pathophysiological functions of ribosomal UFMylation remain unknown. In this study, we systematically demonstrate the significance of UFMylation in the differentiation and maturation of hepatocytes using human embryonic stem cell-derived hepatocyte-like cells and liver bud organoids as experimental platforms. We also develop a strategy to identify UFMylated substrates and confirm that RPL26 is a substrate in the liver. Additionally, we discover that mice with the Rpl26 c.395A>G (p.K132R) mutation are more susceptible to steatosis induced by a high-fat diet. Further investigations reveal a key role of CDK5RAP3 and RPL26 UFMylation in regulating ribosome dissociation. Our findings suggest that ribosome UFMylation serves as an important safeguard for liver development and homeostasis and may represent a potential therapeutic target for nonalcoholic fatty liver disease.
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series Cell Reports
spelling doaj-art-9f31ccb04f6e4fa288cb14023dc0c8e92025-08-20T02:15:06ZengElsevierCell Reports2211-12472025-05-0144511568610.1016/j.celrep.2025.115686UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociationShuchun Yang0Li Wang1Ran Gao2Yanchang Li3Duo Zhang4Chenxi Wang5Guang Liu6Jie Na7Ping Xu8Xiaoyue Wang9Yuyan Jia10Yue Huang11State Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaState Key Laboratory of Medical Proteomics, Beijng Proteome Research Center, National Center for Protein Sciences (Beijing), Research Unit of Proteomics & Research and Development of New Drugs of Chinese Academy of Medical Sciences, Institute of Lifeomics, Beijing 102206, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaCenter for Stem Cell Biology and Regenerative Medicine, School of Medicine, Tsinghua University, Beijing, ChinaState Key Laboratory of Medical Proteomics, Beijng Proteome Research Center, National Center for Protein Sciences (Beijing), Research Unit of Proteomics & Research and Development of New Drugs of Chinese Academy of Medical Sciences, Institute of Lifeomics, Beijing 102206, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, ChinaState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Corresponding authorState Key Laboratory of Common Mechanism Research for Major Disease, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Department of Medical Genetics, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China; Corresponding authorSummary: Ribosomal UFMylation contributes to ribosome heterogeneity and is associated with ribosome-associated quality control at the endoplasmic reticulum. However, the specific pathophysiological functions of ribosomal UFMylation remain unknown. In this study, we systematically demonstrate the significance of UFMylation in the differentiation and maturation of hepatocytes using human embryonic stem cell-derived hepatocyte-like cells and liver bud organoids as experimental platforms. We also develop a strategy to identify UFMylated substrates and confirm that RPL26 is a substrate in the liver. Additionally, we discover that mice with the Rpl26 c.395A>G (p.K132R) mutation are more susceptible to steatosis induced by a high-fat diet. Further investigations reveal a key role of CDK5RAP3 and RPL26 UFMylation in regulating ribosome dissociation. Our findings suggest that ribosome UFMylation serves as an important safeguard for liver development and homeostasis and may represent a potential therapeutic target for nonalcoholic fatty liver disease.http://www.sciencedirect.com/science/article/pii/S2211124725004577CP: MetabolismCP: Molecular biology
spellingShingle Shuchun Yang
Li Wang
Ran Gao
Yanchang Li
Duo Zhang
Chenxi Wang
Guang Liu
Jie Na
Ping Xu
Xiaoyue Wang
Yuyan Jia
Yue Huang
UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
Cell Reports
CP: Metabolism
CP: Molecular biology
title UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
title_full UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
title_fullStr UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
title_full_unstemmed UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
title_short UFMylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
title_sort ufmylation safeguards human hepatocyte differentiation and liver homeostasis by regulating ribosome dissociation
topic CP: Metabolism
CP: Molecular biology
url http://www.sciencedirect.com/science/article/pii/S2211124725004577
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