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Myeloid OTULIN deficiency couples RIPK3-dependent cell death to Nlrp3 inflammasome activation and IL-1β secretion

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Abstract
Loss-of-function mutations in the deubiquitinase OTULIN result in an inflammatory pathology termed "OTULIN-related autoinflammatory syndrome" (ORAS). Genetic mouse models revealed essential roles for OTULIN in inflammatory and cell death signaling, but the mechanisms by which OTULIN deficiency connects cell death to inflammation remain unclear. Here, we identify OTULIN deficiency as a cellular condition that licenses RIPK3-mediated cell death in murine macrophages, leading to Nlrp3 inflammasome activation and subsequent IL-1β secretion. OTULIN deficiency uncoupled Nlrp3 inflammasome activation from gasdermin D-mediated pyroptosis, instead allowing RIPK3-dependent cell death to act as an Nlrp3 inflammasome activator and mechanism for IL-1β release. Accordingly, elevated serum IL-1β levels in myeloid-specific OTULIN-deficient mice were diminished by deleting either Ripk3 or Nlrp3. These findings identify OTULIN as an inhibitor of RIPK3-mediated IL-1β release in mice.

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Citation

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MLA
Doglio, Maria Giulia, et al. “Myeloid OTULIN Deficiency Couples RIPK3-Dependent Cell Death to Nlrp3 Inflammasome Activation and IL-1β Secretion.” SCIENCE IMMUNOLOGY, vol. 8, no. 89, 2023, doi:10.1126/sciimmunol.adf4404.
APA
Doglio, M. G., Verboom, L., Ruilova Sosoranga, E., Frising, U., Asaoka, T., Gansemans, Y., … Wullaert, A. (2023). Myeloid OTULIN deficiency couples RIPK3-dependent cell death to Nlrp3 inflammasome activation and IL-1β secretion. SCIENCE IMMUNOLOGY, 8(89). https://doi.org/10.1126/sciimmunol.adf4404
Chicago author-date
Doglio, Maria Giulia, Lien Verboom, Emily Ruilova Sosoranga, Ulrika Frising, Tomoko Asaoka, Yannick Gansemans, Filip Van Nieuwerburgh, Geert van Loo, and Andy Wullaert. 2023. “Myeloid OTULIN Deficiency Couples RIPK3-Dependent Cell Death to Nlrp3 Inflammasome Activation and IL-1β Secretion.” SCIENCE IMMUNOLOGY 8 (89). https://doi.org/10.1126/sciimmunol.adf4404.
Chicago author-date (all authors)
Doglio, Maria Giulia, Lien Verboom, Emily Ruilova Sosoranga, Ulrika Frising, Tomoko Asaoka, Yannick Gansemans, Filip Van Nieuwerburgh, Geert van Loo, and Andy Wullaert. 2023. “Myeloid OTULIN Deficiency Couples RIPK3-Dependent Cell Death to Nlrp3 Inflammasome Activation and IL-1β Secretion.” SCIENCE IMMUNOLOGY 8 (89). doi:10.1126/sciimmunol.adf4404.
Vancouver
1.
Doglio MG, Verboom L, Ruilova Sosoranga E, Frising U, Asaoka T, Gansemans Y, et al. Myeloid OTULIN deficiency couples RIPK3-dependent cell death to Nlrp3 inflammasome activation and IL-1β secretion. SCIENCE IMMUNOLOGY. 2023;8(89).
IEEE
[1]
M. G. Doglio et al., “Myeloid OTULIN deficiency couples RIPK3-dependent cell death to Nlrp3 inflammasome activation and IL-1β secretion,” SCIENCE IMMUNOLOGY, vol. 8, no. 89, 2023.
@article{01HKMJVGN4GXV8GNSXB6J7RZHY,
  abstract     = {{Loss-of-function mutations in the deubiquitinase OTULIN result in an inflammatory pathology termed "OTULIN-related autoinflammatory syndrome" (ORAS). Genetic mouse models revealed essential roles for OTULIN in inflammatory and cell death signaling, but the mechanisms by which OTULIN deficiency connects cell death to inflammation remain unclear. Here, we identify OTULIN deficiency as a cellular condition that licenses RIPK3-mediated cell death in murine macrophages, leading to Nlrp3 inflammasome activation and subsequent IL-1β secretion. OTULIN deficiency uncoupled Nlrp3 inflammasome activation from gasdermin D-mediated pyroptosis, instead allowing RIPK3-dependent cell death to act as an Nlrp3 inflammasome activator and mechanism for IL-1β release. Accordingly, elevated serum IL-1β levels in myeloid-specific OTULIN-deficient mice were diminished by deleting either Ripk3 or Nlrp3. These findings identify OTULIN as an inhibitor of RIPK3-mediated IL-1β release in mice.}},
  articleno    = {{eadf4404}},
  author       = {{Doglio, Maria Giulia and Verboom, Lien and Ruilova Sosoranga, Emily and Frising, Ulrika and Asaoka, Tomoko and Gansemans, Yannick and Van Nieuwerburgh, Filip and van Loo, Geert and Wullaert, Andy}},
  issn         = {{2470-9468}},
  journal      = {{SCIENCE IMMUNOLOGY}},
  language     = {{eng}},
  number       = {{89}},
  pages        = {{17}},
  title        = {{Myeloid OTULIN deficiency couples RIPK3-dependent cell death to Nlrp3 inflammasome activation and IL-1β secretion}},
  url          = {{http://doi.org/10.1126/sciimmunol.adf4404}},
  volume       = {{8}},
  year         = {{2023}},
}

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