Concurrent remission of lymphoma and Sjögren's disease following anti-CD19 chimeric antigen receptor-T cell therapy for diffuse large B-cell lymphoma: a case report

Front Immunol. 2023 Dec 19:14:1298815. doi: 10.3389/fimmu.2023.1298815. eCollection 2023.

Abstract

Anti-CD19 chimeric antigen receptor (CAR)-T cells not only target CD19-positive malignant lymphoma cells but also normal B cells. The utility of CAR-T cell therapy has been reported in rheumatoid arthritis and systemic lupus erythematosus; however, its use in Sjögren's disease (SjD) remains unknown. In this study, we describe the case of a 76-year-old woman with active SjD for 10 years who was diagnosed with diffuse large B-cell lymphoma. After receiving anti-CD19 CAR-T cell therapy, she achieved complete remission (CR) on day 28. Since the onset of her 10-year history with SjD, she was negative for antinuclear antibodies and anti-Ro-52 for the first time on day 90 after CAR-T cell therapy. Six months after CAR-T cell therapy, the CR status was maintained, serum cytokine levels returned to their normal levels, and dry mouth symptoms improved. The EULAR Sjögren's Syndrome Disease Activity Index score decreased from 5 to 2, indicating a partial remission of SjD activity compared with that before CAR-T cell treatment. In the early stage of treatment, she presented with grade 2 cytokine release syndrome and grade 1 neurotoxicity, which were completely controlled after an active intervention. This case highlights the potential application of CAR-T cells in treating autoimmune diseases, such as SjD.

Keywords: CAR-T cell therapy; Sjögren’s disease; autoimmune diseases; chimeric T cell receptors; immunotherapy.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Antigens, CD19
  • Cell- and Tissue-Based Therapy
  • Female
  • Humans
  • Immunotherapy, Adoptive
  • Lymphoma, Large B-Cell, Diffuse* / therapy
  • Receptors, Chimeric Antigen*
  • Sjogren's Syndrome* / complications
  • Sjogren's Syndrome* / therapy

Substances

  • Receptors, Chimeric Antigen
  • Antigens, CD19

Grants and funding

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This work was supported by National Natural Science Foundation of China [grant number: 82130004, 81830007, 82070204, and 81670176]; Clinical Research Plan of Shanghai Hospital Development Center [grant number: SHDC2020CR1032B]; Shanghai Municipal Education Commission Gaofeng Clinical Medicine [grant number: 20152206 and 20152208]; Multicenter Clinical Research Project by Shanghai JiaoTong University School of Medicine [grant number: DLY201601]; and Chang Jiang Scholars Program and Samuel Waxman Cancer Research Foundation [grant number: N/A].