Characterization of peripheral blood T follicular helper (TFH) cells in patients with type 1 Gaucher disease and carriers

Blood Cells Mol Dis. 2023 May:100:102728. doi: 10.1016/j.bcmd.2023.102728. Epub 2023 Feb 2.

Abstract

Background: Gaucher disease (GD) is the most common autosomal recessive lipid storage disease. In this study, the changes in TFH cells and IL-4 and IL-21 cytokines in blood samples of GD patients, carriers and healthy volunteers were investigated.

Methods: Two pretreatment type 1 GD patients, 20 currently treated type 1 GD patients, 6 carriers, and 27 healthy volunteers were enrolled in the study. TFH cell (CD45RA-CD4+CXCR5+) number, phenotype (PD1, ICOS expression), and cytokine production (IL-21, IL-4) were assessed via flow cytometric assays.

Results: No significant differences were found between the groups with respect to the number, frequency and PD1 or ICOS expression of TFH cells between healthy controls, patients and carriers. However, IL-4+ TFH cells were significantly reduced both in percent and number in the treated GD patients compared with healthy controls (p < 0.05). Interestingly, the IL-21+ TFH cell number was increased in treated GD patients. When TFH cells were examined based on CXCR3 expression, the frequency of the PD1+Th17-Th2-like fraction (CXCR3-) was found to be significantly increased in treated GD patients.

Conclusion: To our knowledge, this is the first study to assess TFH cells in GD patients, and to show that the production of IL-4 and IL-21 by TFH cells and their subsets may be altered in type 1 GD patients.

Keywords: Gaucher disease; IL-21; IL-4; TFH cells.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • CD4-Positive T-Lymphocytes
  • Gaucher Disease* / metabolism
  • Humans
  • Interleukin-4
  • Interleukins
  • T Follicular Helper Cells*
  • T-Lymphocytes, Helper-Inducer / metabolism

Substances

  • Interleukin-4
  • Interleukins