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Updated: Nov 8, 2025

Stimulation of Cytoplasmic DNA Sensing Pathways In Vitro and In Vivo
Published on: September 18, 2014
Cellular pyrimidine imbalance triggers mitochondrial DNA-dependent innate immunity
Hans-Georg Sprenger1,2, Thomas MacVicar1, Amir Bahat1
1Max Planck Institute for Biology of Ageing, Cologne, Germany.
Abstract:
Cytosolic mitochondrial DNA (mtDNA) elicits a type I interferon response, but signals triggering the release of mtDNA from mitochondria remain enigmatic. Here, we show that mtDNA-dependent immune signalling via the cyclic GMP-AMP synthase‒stimulator of interferon genes‒TANK-binding kinase 1 (cGAS-STING-TBK1) pathway is under metabolic control and is induced by cellular pyrimidine deficiency. The mitochondrial protease YME1L preserves pyrimidine pools by supporting de novo nucleotide synthesis and by proteolysis of the pyrimidine nucleotide carrier SLC25A33. Deficiency of YME1L causes inflammation in mouse retinas and in cultured cells. It drives the release of mtDNA and a cGAS-STING-TBK1-dependent inflammatory response, which requires SLC25A33 and is suppressed upon replenishment of cellular pyrimidine pools. Overexpression of SLC25A33 is sufficient to induce immune signalling by mtDNA. Similarly, depletion of cytosolic nucleotides upon inhibition of de novo pyrimidine synthesis triggers mtDNA-dependent immune responses in wild-type cells. Our results thus identify mtDNA release and innate immune signalling as a metabolic response to cellular pyrimidine deficiencies.
Insights
Cellular pyrimidine deficiency triggers mitochondrial DNA release and innate immune responses. This metabolic control involves the YME1L protease and the cGAS-STING-TBK1 pathway, revealing a link between metabolism and immunity.
Area of Science:
- Immunology
- Cell Biology
- Metabolic Regulation
Background:
- Cytosolic mitochondrial DNA (mtDNA) activates type I interferon responses, but triggers for mtDNA release are unknown.
- The cyclic GMP-AMP synthase‒stimulator of interferon genes‒TANK-binding kinase 1 (cGAS-STING-TBK1) pathway mediates mtDNA-dependent immune signaling.
Purpose of the Study:
- To investigate the metabolic control of mtDNA release and subsequent innate immune signaling.
- To identify the molecular mechanisms linking cellular metabolism to immune activation via cytosolic mtDNA.
Main Methods:
- Investigated the role of the mitochondrial protease YME1L in pyrimidine metabolism and mtDNA release.
- Utilized cell culture and mouse models to study inflammation in response to YME1L deficiency.
- Examined the involvement of the cGAS-STING-TBK1 pathway and SLC25A33 in mtDNA-induced immune responses.
- Assessed the impact of pyrimidine pool replenishment on immune signaling.
Main Results:
- YME1L deficiency leads to pyrimidine depletion, causing mtDNA release and cGAS-STING-TBK1-dependent inflammation in mouse retinas and cell cultures.
- The immune response requires SLC25A33 and is suppressed by restoring pyrimidine levels.
- Overexpression of SLC25A33 or inhibition of de novo pyrimidine synthesis induces mtDNA-dependent immune signaling.
Conclusions:
- Cellular pyrimidine deficiency is a key trigger for mtDNA release and innate immune activation.
- The YME1L-SLC25A33 axis plays a crucial role in maintaining pyrimidine homeostasis and preventing aberrant immune responses.
- mtDNA release and subsequent immune signaling represent a metabolic response to pyrimidine shortages.
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