Genetically encoded Runx3 and CD4+ intestinal epithelial lymphocyte deficiencies link SKG mouse and human predisposition to spondyloarthropathy

Genetically encoded Runx3 and CD4+ intestinal epithelial lymphocyte deficiencies link SKG mouse and human predisposition to spondyloarthropathy

Copyright © 2022. Published by Elsevier Inc.

Détails bibliographiques
Publié dans:Clinical immunology (Orlando, Fla.). - 1999. - 247(2023) vom: 15. Feb., Seite 109220
Auteur principal: Bhuyan, Zaied Ahmed (Auteur)
Autres auteurs: Rahman, M Arifur, Maradana, Muralidhara Rao, Mehdi, Ahmed M, Bergot, Anne-Sophie, Simone, Davide, El-Kurdi, Marya, Garrido-Mesa, Jose, Cai, Cheng Bang Benjamin, Cameron, Amy J, Hanson, Aimee L, Nel, Hendrik J, Kenna, Tony, Leo, Paul, Rehaume, Linda, Brown, Matthew A, Ciccia, Francesco, Thomas, Ranjeny
Format: Article en ligne
Langue:English
Publié: 2023
Accès à la collection:Clinical immunology (Orlando, Fla.)
Sujets:Journal Article Research Support, Non-U.S. Gov't Ankylosing spondylitis Immunodeficiency Intestine Runx3 T cells Core Binding Factor Alpha 3 Subunit Receptors, Antigen, T-Cell, alpha-beta Runx3 protein, human plus... Transforming Growth Factor beta Runx3 protein, mouse
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520 |a Disturbances in immune regulation, intestinal dysbiosis and inflammation characterize ankylosing spondylitis (AS), which is associated with RUNX3 loss-of-function variants. ZAP70W163C mutant (SKG) mice have reduced ZAP70 signaling, spondyloarthritis and ileitis. In small intestine, Foxp3+ regulatory T cells (Treg) and CD4+CD8αα+TCRαβ+ intraepithelial lymphocytes (CD4-IEL) control inflammation. TGF-β and retinoic acid (RA)-producing dendritic cells and MHC-class II+ intestinal epithelial cells (IEC) are required for Treg and CD4-IEL differentiation from CD4+ conventional or Treg precursors, with upregulation of Runx3 and suppression of ThPOK. We show in SKG mouse ileum, that ZAP70W163C or ZAP70 inhibition prevented CD4-IEL but not Treg differentiation, dysregulating Runx3 and ThPOK. TGF-β/RA-mediated CD4-IEL development, T-cell IFN-γ production, MHC class-II+ IEC, tissue-resident memory T-cell and Runx3-regulated genes were reduced. In AS intestine, CD4-IEL were decreased, while in AS blood CD4+CD8+ T cells were reduced and Treg increased. Thus, genetically-encoded TCR signaling dysfunction links intestinal T-cell immunodeficiency in mouse and human spondyloarthropathy 
650 4 |a Journal Article 
650 4 |a Research Support, Non-U.S. Gov't 
650 4 |a Ankylosing spondylitis 
650 4 |a Immunodeficiency 
650 4 |a Intestine 
650 4 |a Runx3 
650 4 |a T cells 
650 7 |a Core Binding Factor Alpha 3 Subunit  |2 NLM 
650 7 |a Receptors, Antigen, T-Cell, alpha-beta  |2 NLM 
650 7 |a Runx3 protein, human  |2 NLM 
650 7 |a Transforming Growth Factor beta  |2 NLM 
650 7 |a Runx3 protein, mouse  |2 NLM 
700 1 |a Rahman, M Arifur  |e verfasserin  |4 aut 
700 1 |a Maradana, Muralidhara Rao  |e verfasserin  |4 aut 
700 1 |a Mehdi, Ahmed M  |e verfasserin  |4 aut 
700 1 |a Bergot, Anne-Sophie  |e verfasserin  |4 aut 
700 1 |a Simone, Davide  |e verfasserin  |4 aut 
700 1 |a El-Kurdi, Marya  |e verfasserin  |4 aut 
700 1 |a Garrido-Mesa, Jose  |e verfasserin  |4 aut 
700 1 |a Cai, Cheng Bang Benjamin  |e verfasserin  |4 aut 
700 1 |a Cameron, Amy J  |e verfasserin  |4 aut 
700 1 |a Hanson, Aimee L  |e verfasserin  |4 aut 
700 1 |a Nel, Hendrik J  |e verfasserin  |4 aut 
700 1 |a Kenna, Tony  |e verfasserin  |4 aut 
700 1 |a Leo, Paul  |e verfasserin  |4 aut 
700 1 |a Rehaume, Linda  |e verfasserin  |4 aut 
700 1 |a Brown, Matthew A  |e verfasserin  |4 aut 
700 1 |a Ciccia, Francesco  |e verfasserin  |4 aut 
700 1 |a Thomas, Ranjeny  |e verfasserin  |4 aut 
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