Deficiency of CBL and CBLB ubiquitin ligases leads to hyper T follicular helper cell responses and lupus by reducing BCL6 degradation

Immunity. 2024 Jul 9;57(7):1603-1617.e7. doi: 10.1016/j.immuni.2024.04.023. Epub 2024 May 17.

Abstract

Recent evidence reveals hyper T follicular helper (Tfh) cell responses in systemic lupus erythematosus (SLE); however, molecular mechanisms responsible for hyper Tfh cell responses and whether they cause SLE are unclear. We found that SLE patients downregulated both ubiquitin ligases, casitas B-lineage lymphoma (CBL) and CBLB (CBLs), in CD4+ T cells. T cell-specific CBLs-deficient mice developed hyper Tfh cell responses and SLE, whereas blockade of Tfh cell development in the mutant mice was sufficient to prevent SLE. ICOS was upregulated in SLE Tfh cells, whose signaling increased BCL6 by attenuating BCL6 degradation via chaperone-mediated autophagy (CMA). Conversely, CBLs restrained BCL6 expression by ubiquitinating ICOS. Blockade of BCL6 degradation was sufficient to enhance Tfh cell responses. Thus, the compromised expression of CBLs is a prevalent risk trait shared by SLE patients and causative to hyper Tfh cell responses and SLE. The ICOS-CBLs axis may be a target to treat SLE.

Keywords: BCL6; CBL family proteins; CMA; ICOS; SLE; Tfh; chaperone-mediated autophagy; follicular helper T cell; systemic lupus erythematosus; ubiquitination.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptor Proteins, Signal Transducing* / genetics
  • Adaptor Proteins, Signal Transducing* / metabolism
  • Animals
  • Autophagy / immunology
  • Female
  • Humans
  • Inducible T-Cell Co-Stimulator Protein* / genetics
  • Inducible T-Cell Co-Stimulator Protein* / metabolism
  • Lupus Erythematosus, Systemic* / genetics
  • Lupus Erythematosus, Systemic* / immunology
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout*
  • Proteolysis
  • Proto-Oncogene Proteins c-bcl-6* / genetics
  • Proto-Oncogene Proteins c-bcl-6* / metabolism
  • Proto-Oncogene Proteins c-cbl* / deficiency
  • Proto-Oncogene Proteins c-cbl* / genetics
  • Proto-Oncogene Proteins c-cbl* / metabolism
  • Signal Transduction / immunology
  • T Follicular Helper Cells* / immunology
  • T-Lymphocytes, Helper-Inducer / immunology
  • Ubiquitination

Substances

  • Adaptor Proteins, Signal Transducing
  • BCL6 protein, human
  • Bcl6 protein, mouse
  • Cbl protein, mouse
  • CBLB protein, human
  • Cblb protein, mouse
  • Icos protein, mouse
  • Inducible T-Cell Co-Stimulator Protein
  • Proto-Oncogene Proteins c-bcl-6
  • Proto-Oncogene Proteins c-cbl
  • CBL protein, human