Interleukin-21 restores immunoglobulin production ex vivo in patients with common variable immunodeficiency and selective IgA deficiency

Blood. 2009 Nov 5;114(19):4089-98. doi: 10.1182/blood-2009-02-207423. Epub 2009 Sep 8.

Abstract

Interleukin-21 (IL-21) is an important promoter for differentiation of human B cells into immunoglobulin (Ig)-secreting cells. The objective of this study was to evaluate an IL-21-based approach to induce immunoglobulin production in B cells from patients with common variable immunodeficiency (CVID) or selective IgA deficiency (IgAD). We show that a combination of IL-21, IL-4, and anti-CD40 stimulation induces class-switch recombination to IgG and IgA and differentiation of Ig-secreting cells, consisting of both surface IgG(+) (sIgG(+)) and sIgA(+) B cells and CD138(+) plasma cells, in patients with CVID or IgAD. Stimulation with IL-21 was far more effective than stimulation with IL-4 or IL-10. Moreover, spontaneous apoptosis of CD19(+) B cells from patients with CVID or IgAD was prevented by a combination of IL-21, IL-4, and anti-CD40 stimulation. Analysis of IL-21 and IL-21 receptor (IL-21R) mRNA expression upon anti-CD3 stimulation of T cells, however, showed no evidence for defective IL-21 expression in CVID patients and sequencing of the coding regions of the IL21 gene did not reveal any mutations, suggesting a regulatory defect. Thus, our work provides an initial basis for a potential therapeutic role of IL-21 to reconstitute immunoglobulin production in CVID and IgAD.

MeSH terms

  • Apoptosis / drug effects
  • B-Lymphocytes / drug effects
  • B-Lymphocytes / immunology
  • B-Lymphocytes / pathology
  • Case-Control Studies
  • Cell Differentiation / drug effects
  • Common Variable Immunodeficiency / drug therapy*
  • Common Variable Immunodeficiency / genetics
  • Common Variable Immunodeficiency / immunology*
  • Drug Synergism
  • Gene Expression
  • Humans
  • IgA Deficiency / drug therapy*
  • IgA Deficiency / genetics
  • IgA Deficiency / immunology*
  • Immunoglobulin A / biosynthesis
  • Immunoglobulin Class Switching / drug effects
  • Immunoglobulin G / biosynthesis
  • Immunoglobulins / biosynthesis*
  • In Vitro Techniques
  • Interleukin-10 / pharmacology
  • Interleukin-2 / administration & dosage
  • Interleukin-2 / pharmacology
  • Interleukin-21 Receptor alpha Subunit / genetics
  • Interleukin-4 / administration & dosage
  • Interleukin-4 / pharmacology
  • Interleukins / administration & dosage
  • Interleukins / genetics
  • Interleukins / pharmacology*
  • RNA, Messenger / genetics
  • RNA, Messenger / metabolism
  • Recombinant Proteins / administration & dosage
  • Recombinant Proteins / pharmacology

Substances

  • IL10 protein, human
  • IL2 protein, human
  • IL21R protein, human
  • IL4 protein, human
  • Immunoglobulin A
  • Immunoglobulin G
  • Immunoglobulins
  • Interleukin-2
  • Interleukin-21 Receptor alpha Subunit
  • Interleukins
  • RNA, Messenger
  • Recombinant Proteins
  • Interleukin-10
  • Interleukin-4
  • interleukin-21