Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions

Abstract Most mitochondrial proteins encoded in the nuclear genome are synthesized in the cytoplasm. These proteins subsequently undergo maturation through the cleavage of a signal sequence at the N-terminus by one or two mitochondrial signal peptidases, which is essential for their function within...

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Main Authors: Yuka Nozaki, Masaki Kobayashi, Tomoyoshi Fukuoh, Mamiko Ishimatsu, Takumi Narita, Kanari Taki, Yuto Hirao, Shota Ayabe, Miku Yokoyama, Yuina Otani, Yuhei Mizunoe, Mami Matsumoto, Nobuhiko Ohno, Tomonori Kaifu, Shogo Okazaki, Ryo Goitsuka, Yoshimi Nakagawa, Hitoshi Shimano, Yoichiro Iwakura, Yoshikazu Higami
Format: Article
Language:English
Published: Nature Portfolio 2025-04-01
Series:Scientific Reports
Online Access:https://doi.org/10.1038/s41598-025-97307-6
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author Yuka Nozaki
Masaki Kobayashi
Tomoyoshi Fukuoh
Mamiko Ishimatsu
Takumi Narita
Kanari Taki
Yuto Hirao
Shota Ayabe
Miku Yokoyama
Yuina Otani
Yuhei Mizunoe
Mami Matsumoto
Nobuhiko Ohno
Tomonori Kaifu
Shogo Okazaki
Ryo Goitsuka
Yoshimi Nakagawa
Hitoshi Shimano
Yoichiro Iwakura
Yoshikazu Higami
author_facet Yuka Nozaki
Masaki Kobayashi
Tomoyoshi Fukuoh
Mamiko Ishimatsu
Takumi Narita
Kanari Taki
Yuto Hirao
Shota Ayabe
Miku Yokoyama
Yuina Otani
Yuhei Mizunoe
Mami Matsumoto
Nobuhiko Ohno
Tomonori Kaifu
Shogo Okazaki
Ryo Goitsuka
Yoshimi Nakagawa
Hitoshi Shimano
Yoichiro Iwakura
Yoshikazu Higami
author_sort Yuka Nozaki
collection DOAJ
description Abstract Most mitochondrial proteins encoded in the nuclear genome are synthesized in the cytoplasm. These proteins subsequently undergo maturation through the cleavage of a signal sequence at the N-terminus by one or two mitochondrial signal peptidases, which is essential for their function within mitochondria. The present study demonstrates that adipocyte-specific knockout of one mitochondrial signal peptidase, mitochondrial intermediate peptidase (MIPEP), resulted in disordered mitochondrial proteostasis of MIPEP substrate proteins and their defective maturation. MIPEP deficiency in white and brown adipocytes suppressed the expression of adipocyte differentiation, lipid metabolism, and mitochondrial biogenesis genes. These alterations led to lipoatrophy in white adipose tissue and the whitening of brown adipose tissue. Additionally, it induced an atypical mitochondrial unfolded protein response and local inflammation in white and brown adipose tissue. Furthermore, it induced fatty liver and splenomegaly and caused systemic impairments in glucose metabolism and inflammation. These findings indicate that maturation defects of certain mitochondrial proteins and subsequent proteostasis disorders in white and brown adipocytes cause chronic and systemic inflammatory and metabolic dysfunctions.
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spelling doaj-art-07ca8f617fce4cddace436454056357d2025-08-20T02:17:50ZengNature PortfolioScientific Reports2045-23222025-04-0115111910.1038/s41598-025-97307-6Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctionsYuka Nozaki0Masaki Kobayashi1Tomoyoshi Fukuoh2Mamiko Ishimatsu3Takumi Narita4Kanari Taki5Yuto Hirao6Shota Ayabe7Miku Yokoyama8Yuina Otani9Yuhei Mizunoe10Mami Matsumoto11Nobuhiko Ohno12Tomonori Kaifu13Shogo Okazaki14Ryo Goitsuka15Yoshimi Nakagawa16Hitoshi Shimano17Yoichiro Iwakura18Yoshikazu Higami19Laboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceSection of Electron Microscopy, Supportive Center for Brain Research, National Institute for Physiological SciencesDepartment of Anatomy, Division of Histology and Cell Biology, School of Medicine, Jichi Medical UniversityDivision of Immunology, Faculty of Medicine, Tohoku Medical and Pharmaceutical UniversityResearch Institute for Biomedical Sciences (RIBS), Tokyo University of ScienceResearch Institute for Biomedical Sciences (RIBS), Tokyo University of ScienceDivision of Complex Biosystem Research, Department of Research and Development, Institute of Natural Medicine, University of ToyamaDepartment of Endocrinology and Metabolism, Institute of Medicine, University of TsukubaResearch Institute for Biomedical Sciences (RIBS), Tokyo University of ScienceLaboratory of Molecular Pathology and Metabolic Disease, Faculty of Pharmaceutical Sciences, Tokyo University of ScienceAbstract Most mitochondrial proteins encoded in the nuclear genome are synthesized in the cytoplasm. These proteins subsequently undergo maturation through the cleavage of a signal sequence at the N-terminus by one or two mitochondrial signal peptidases, which is essential for their function within mitochondria. The present study demonstrates that adipocyte-specific knockout of one mitochondrial signal peptidase, mitochondrial intermediate peptidase (MIPEP), resulted in disordered mitochondrial proteostasis of MIPEP substrate proteins and their defective maturation. MIPEP deficiency in white and brown adipocytes suppressed the expression of adipocyte differentiation, lipid metabolism, and mitochondrial biogenesis genes. These alterations led to lipoatrophy in white adipose tissue and the whitening of brown adipose tissue. Additionally, it induced an atypical mitochondrial unfolded protein response and local inflammation in white and brown adipose tissue. Furthermore, it induced fatty liver and splenomegaly and caused systemic impairments in glucose metabolism and inflammation. These findings indicate that maturation defects of certain mitochondrial proteins and subsequent proteostasis disorders in white and brown adipocytes cause chronic and systemic inflammatory and metabolic dysfunctions.https://doi.org/10.1038/s41598-025-97307-6
spellingShingle Yuka Nozaki
Masaki Kobayashi
Tomoyoshi Fukuoh
Mamiko Ishimatsu
Takumi Narita
Kanari Taki
Yuto Hirao
Shota Ayabe
Miku Yokoyama
Yuina Otani
Yuhei Mizunoe
Mami Matsumoto
Nobuhiko Ohno
Tomonori Kaifu
Shogo Okazaki
Ryo Goitsuka
Yoshimi Nakagawa
Hitoshi Shimano
Yoichiro Iwakura
Yoshikazu Higami
Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
Scientific Reports
title Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
title_full Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
title_fullStr Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
title_full_unstemmed Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
title_short Mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
title_sort mipep deficiency in adipocytes impairs mitochondrial protein maturation and leads to systemic inflammation and metabolic dysfunctions
url https://doi.org/10.1038/s41598-025-97307-6
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