Abstract
Germline telomere maintenance defects are associated with an increased incidence of inflammatory diseases in humans, yet whether and how telomere dysfunction causes inflammation are not known. Here, we show that telomere dysfunction drives pATM/c-ABL-mediated activation of the YAP1 transcription factor, up-regulating the major pro-inflammatory factor, pro-IL-18. The colonic microbiome stimulates cytosolic receptors activating caspase-1 which cleaves pro-IL-18 into mature IL-18, leading to recruitment of interferon (IFN)-γ-secreting T cells and intestinal inflammation. Correspondingly, patients with germline telomere maintenance defects exhibit DNA damage (γH2AX) signaling together with elevated YAP1 and IL-18 expression. In mice with telomere dysfunction, telomerase reactivation in the intestinal epithelium or pharmacological inhibition of ATM, YAP1, or caspase-1 as well as antibiotic treatment, dramatically reduces IL-18 and intestinal inflammation. Thus, telomere dysfunction-induced activation of the ATM-YAP1-pro-IL-18 pathway in epithelium is a key instigator of tissue inflammation.
Publication types
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Research Support, N.I.H., Extramural
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Research Support, Non-U.S. Gov't
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Research Support, U.S. Gov't, Non-P.H.S.
MeSH terms
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Adaptor Proteins, Signal Transducing / antagonists & inhibitors
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Adaptor Proteins, Signal Transducing / genetics
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Adaptor Proteins, Signal Transducing / metabolism*
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Animals
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Anti-Bacterial Agents / therapeutic use
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Ataxia Telangiectasia Mutated Proteins / antagonists & inhibitors
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Ataxia Telangiectasia Mutated Proteins / metabolism
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Caspase 1 / metabolism
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Cell Cycle Proteins / antagonists & inhibitors
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Cell Cycle Proteins / genetics
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Cell Cycle Proteins / metabolism*
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Child
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Colon / metabolism
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Colon / microbiology
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Colon / pathology
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Gastrointestinal Diseases / pathology
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Gastrointestinal Microbiome / drug effects
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Gastrointestinal Microbiome / physiology
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Humans
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Inflammation / drug therapy
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Inflammation / metabolism
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Inflammation / microbiology
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Inflammation / pathology*
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Interleukin-18 / genetics
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Interleukin-18 / metabolism
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Intestinal Mucosa / metabolism
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Intestinal Mucosa / pathology
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Mice
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Mice, Mutant Strains
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Phosphorylation
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Protein Precursors / genetics
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Protein Precursors / metabolism
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Signal Transduction
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Telomerase / genetics
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Telomerase / metabolism
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Telomere / pathology*
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YAP-Signaling Proteins
Substances
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Adaptor Proteins, Signal Transducing
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Anti-Bacterial Agents
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Cell Cycle Proteins
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Interleukin-18
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Protein Precursors
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YAP-Signaling Proteins
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Yap1 protein, mouse
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Ataxia Telangiectasia Mutated Proteins
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Atm protein, mouse
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Telomerase
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Tert protein, mouse
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Casp1 protein, mouse
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Caspase 1