TY - JOUR
T1 - IL-15, gluten and HLA-DQ8 drive tissue destruction in coeliac disease
AU - Abadie, Valérie
AU - Kim, Sangman M.
AU - Lejeune, Thomas
AU - Palanski, Brad A.
AU - Ernest, Jordan D.
AU - Tastet, Olivier
AU - Voisine, Jordan
AU - Discepolo, Valentina
AU - Marietta, Eric V.
AU - Hawash, Mohamed B.F.
AU - Ciszewski, Cezary
AU - Bouziat, Romain
AU - Panigrahi, Kaushik
AU - Horwath, Irina
AU - Zurenski, Matthew A.
AU - Lawrence, Ian
AU - Dumaine, Anne
AU - Yotova, Vania
AU - Grenier, Jean Christophe
AU - Murray, Joseph A.
AU - Khosla, Chaitan
AU - Barreiro, Luis B.
AU - Jabri, Bana
N1 - Funding Information:
Acknowledgements We thank patients with coeliac disease and their family members, as well as the University of Chicago Celiac Disease Center, for supporting our research. We thank BZ-Histo Services for the histological processing of intestinal samples. We also thank the Human Tissue Resource Center and the Integrated Light Microscopy Core Facility at the University of Chicago. This work was supported by grants from NIH: R01 DK67180 and R01 DK63158, and Digestive Diseases Research Core Center P30 DK42086 at the University of Chicago as well as by funding from F. Oliver Nicklin associated with the First Analysis Institute of Integrative Studies and the Regenstein Foundation to B.J., from the CIHR (catalyst grant in environments,
Funding Information:
genes, and chronic disease) to V.A. and L.B.B., the SickKids Foundation (NI15-040) and the Canadian Celiac Association to V.A., from NIH: R01 DK063158 and R01 DK100619 to C.K., award from the Wallonie-Bruxelles International-World Excellence and from FRQNT to T.L. The Carlino Fellowship for Celiac Disease Research from the University of Chicago Celiac Disease Center supported V.D.
Publisher Copyright:
© 2020, The Author(s), under exclusive licence to Springer Nature Limited.
PY - 2020/2/27
Y1 - 2020/2/27
N2 - Coeliac disease is a complex, polygenic inflammatory enteropathy caused by exposure to dietary gluten that occurs in a subset of genetically susceptible individuals who express either the HLA-DQ8 or HLA-DQ2 haplotypes1,2. The need to develop non-dietary treatments is now widely recognized3, but no pathophysiologically relevant gluten- and HLA-dependent preclinical model exists. Furthermore, although studies in humans have led to major advances in our understanding of the pathogenesis of coeliac disease4, the respective roles of disease-predisposing HLA molecules, and of adaptive and innate immunity in the development of tissue damage, have not been directly demonstrated. Here we describe a mouse model that reproduces the overexpression of interleukin-15 (IL-15) in the gut epithelium and lamina propria that is characteristic of active coeliac disease, expresses the predisposing HLA-DQ8 molecule, and develops villous atrophy after ingestion of gluten. Overexpression of IL-15 in both the epithelium and the lamina propria is required for the development of villous atrophy, which demonstrates the location-dependent central role of IL-15 in the pathogenesis of coeliac disease. In addition, CD4+ T cells and HLA-DQ8 have a crucial role in the licensing of cytotoxic T cells to mediate intestinal epithelial cell lysis. We also demonstrate a role for the cytokine interferon-γ (IFNγ) and the enzyme transglutaminase 2 (TG2) in tissue destruction. By reflecting the complex interaction between gluten, genetics and IL-15-driven tissue inflammation, this mouse model provides the opportunity to both increase our understanding of coeliac disease, and develop new therapeutic strategies.
AB - Coeliac disease is a complex, polygenic inflammatory enteropathy caused by exposure to dietary gluten that occurs in a subset of genetically susceptible individuals who express either the HLA-DQ8 or HLA-DQ2 haplotypes1,2. The need to develop non-dietary treatments is now widely recognized3, but no pathophysiologically relevant gluten- and HLA-dependent preclinical model exists. Furthermore, although studies in humans have led to major advances in our understanding of the pathogenesis of coeliac disease4, the respective roles of disease-predisposing HLA molecules, and of adaptive and innate immunity in the development of tissue damage, have not been directly demonstrated. Here we describe a mouse model that reproduces the overexpression of interleukin-15 (IL-15) in the gut epithelium and lamina propria that is characteristic of active coeliac disease, expresses the predisposing HLA-DQ8 molecule, and develops villous atrophy after ingestion of gluten. Overexpression of IL-15 in both the epithelium and the lamina propria is required for the development of villous atrophy, which demonstrates the location-dependent central role of IL-15 in the pathogenesis of coeliac disease. In addition, CD4+ T cells and HLA-DQ8 have a crucial role in the licensing of cytotoxic T cells to mediate intestinal epithelial cell lysis. We also demonstrate a role for the cytokine interferon-γ (IFNγ) and the enzyme transglutaminase 2 (TG2) in tissue destruction. By reflecting the complex interaction between gluten, genetics and IL-15-driven tissue inflammation, this mouse model provides the opportunity to both increase our understanding of coeliac disease, and develop new therapeutic strategies.
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U2 - 10.1038/s41586-020-2003-8
DO - 10.1038/s41586-020-2003-8
M3 - Article
C2 - 32051586
AN - SCOPUS:85079429551
SN - 0028-0836
VL - 578
SP - 600
EP - 604
JO - Nature
JF - Nature
IS - 7796
ER -