Altered responses to homeostatic cytokines in patients with idiopathic CD4 lymphocytopenia

PLoS One. 2013;8(1):e55570. doi: 10.1371/journal.pone.0055570. Epub 2013 Jan 30.

Abstract

Idiopathic CD4 lymphocytopenia (ICL) is a rare immune deficiency characterized by a protracted CD4(+) T cell loss of unknown etiology and by the occurrence of opportunistic infections similar to those seen in AIDS. We investigated whether a defect in responses to cytokines that control CD4(+) T cell homeostasis could play a role in ICL. Immunophenotype and signaling responses to interleukin-7 (IL-7), IL-2, and thymic stromal lymphopoietin (TSLP) were analyzed by flow cytometry in CD4(+) T cells from 15 ICL patients and 15 healthy blood donors. The induction of phospho-STAT5 after IL-7 stimulation was decreased in memory CD4(+) T cells of some ICL patients, which correlated with a decreased expression of the IL-7Rα receptor chain (R = 0.74, p<0.005) and with lower CD4(+) T cell counts (R = 0.69, p<0.005). IL-2 responses were also impaired, both in the Treg and conventional memory subsets. Decreased IL-2 responses correlated with decreased IL-7 responses (R = 0.75, p<0.005), pointing to combined defects that may significantly perturb CD4(+) T cell homeostasis in a subset of ICL patients. Unexpectedly, responses to the IL-7-related cytokine TSLP were increased in ICL patients, while they remained barely detectable in healthy controls. TSLP responses correlated inversely with IL-7 responses (R = -0.41; p<0.05), suggesting a cross-regulation between the two cytokine systems. In conclusion, IL-7 and IL-2 signaling are impaired in ICL, which may account for the loss of CD4(+) T cell homeostasis. Increased TSLP responses point to a compensatory homeostatic mechanism that may mitigate defects in γc cytokine responses.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • CD4 Lymphocyte Count
  • CD4-Positive T-Lymphocytes / immunology*
  • CD4-Positive T-Lymphocytes / metabolism
  • Cytokines / immunology*
  • Cytokines / metabolism
  • Cytokines / pharmacology
  • Female
  • Homeostasis / immunology*
  • Humans
  • Immunophenotyping
  • Interleukin-2 / immunology
  • Interleukin-2 / metabolism
  • Interleukin-2 / pharmacology
  • Interleukin-2 Receptor alpha Subunit / metabolism
  • Interleukin-7 / immunology
  • Interleukin-7 / metabolism
  • Interleukin-7 / pharmacology
  • Interleukin-7 Receptor alpha Subunit / metabolism
  • Lymphocyte Activation
  • Lymphopenia / immunology*
  • Lymphopenia / metabolism
  • Male
  • Middle Aged
  • Phosphorylation / drug effects
  • STAT5 Transcription Factor / metabolism
  • Thymic Stromal Lymphopoietin
  • Young Adult

Substances

  • Cytokines
  • Interleukin-2
  • Interleukin-2 Receptor alpha Subunit
  • Interleukin-7
  • Interleukin-7 Receptor alpha Subunit
  • STAT5 Transcription Factor
  • Thymic Stromal Lymphopoietin

Grants and funding

I.L. is the recipient of a fellowship from the SENACYT Research Agency from Panama. This work was supported by Assistance Publique des Hpitaux de Paris (AP-HP), Ligue contre le Cancer du Grand Est, and by the Pasteur Institute, Paris. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.