mtDNA leakage promotes neuron–glia crosstalk to induce epilepsy by cGAS–STING-driven neuroinflammation and serine metabolic reprogramming

J Jie Jiang M Meiling Zuo (College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University) K Kehan Zhao (College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University) Z Zhihao Ling (College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University) Z Zhida Wu (College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University) D Dongfang Xue (Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology) S Shouyong Mo (Department of Laboratory Medicine, The Fifth Affiliated Hospital of Wenzhou Medical University) Y Yuanhui Liu (Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology) Y Yongjun Chen (Department of Neurology, The Affiliated Nanhua Hospital, Hengyang Medical School, University of South China) J Jie Wang (State Key Laboratory of Molecular Oncology, Beijing Key Laboratory, CAMS Key Laboratory of Translational Research on Lung Cancer, Department of Medical Oncology Cancer Hospital, Chinese Academy of Medical Sciences Beijing China) B Bin Lu (The University of Tokyo , , ,) C Chuanzhou Li (Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology) Y Yaqi Duan (Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology) H He He (Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology) Z Zhiyin Song (Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology)

Abstract

Epilepsy is increasingly recognized as a disorder involving metabolic dysregulation beyond neural hyperexcitability, yet the underlying metabolic mechanisms remain poorly defined. Here, we identify a mitochondrion–immunity–metabolism axis that drives spontaneous chronic epilepsy. Brain-specific deletion of Mic19 impairs mitochondrial cristae structure and mitochondrial integrity in neurons, leading to activation of the Z-mitochondrial DNA (mtDNA)–ZBP1–RIPK3–mixed lineage kinase domain-like protein (MLKL) axis and p-MLKL-mediated pore formation on the mitochondrial membrane. This process results in cytosolic and extracellular leakage of mtDNA, which is subsequently taken up by microglia and triggers cyclic GMP-AMP synthase (cGAS)–STING-dependent inflammatory signaling. The resulting neuroinflammation promotes sustained activation of astrocytes. Critically, reactive astrocytes undergo profound metabolic reprogramming, marked by upregulated glycolysis and enhanced L-serine biosynthesis. Astrocyte-derived L-serine is subsequently transferred to neurons and converted into D-serine, a key NMDA receptor coagonist that enhances neuronal excitability. This metabolic shift in astrocytes exacerbates excitotoxicity and sustains epileptic activity. Importantly, pharmacologic inhibition of STING with H-151 treatment markedly suppresses seizures, reinforcing the therapeutic potential of targeting immunometabolic crosstalk in epilepsy. Our findings reveal that mtDNA-mediated cGAS–STING activation and D-serine act as important drivers of epilepsy initiation, offering mechanistic insights into neuron–microglia–astrocyte crosstalk and highlighting immunometabolic modulation as a promising therapeutic strategy for epilepsy.

Article Details

Volume / Issue Vol. 123, Issue 9
Published March 03, 2026
ISSN 0027-8424
Publisher National Academy of Sciences

Authors (15)

J

Jie Jiang

M

Meiling Zuo

College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University

K

Kehan Zhao

College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University

Z

Zhihao Ling

College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University

Z

Zhida Wu

College of Life Sciences, Frontier Science Center for Immunology and Metabolism, Wuhan University

D

Dongfang Xue

Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology

S

Shouyong Mo

Department of Laboratory Medicine, The Fifth Affiliated Hospital of Wenzhou Medical University

Y

Yuanhui Liu

Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology

Y

Yongjun Chen

Department of Neurology, The Affiliated Nanhua Hospital, Hengyang Medical School, University of South China

J

Jie Wang

State Key Laboratory of Molecular Oncology, Beijing Key Laboratory, CAMS Key Laboratory of Translational Research on Lung Cancer, Department of Medical Oncology Cancer Hospital, Chinese Academy of Medical Sciences Beijing China

B

Bin Lu

The University of Tokyo , , ,

C

Chuanzhou Li

Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology

Y

Yaqi Duan

Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology

H

He He

Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology

Z

Zhiyin Song

Institute of pathology, Tongji Hospital, School of Basic Medicine, State Key Laboratory for Diagnosis and Treatment of Severe Zoonotic Infectious Diseases, Tongji Medical College, Huazhong University of Science and Technology