Distinct immune trajectories in patients with chromosome 22q11.2 deletion syndrome and immune-mediated diseases

J Allergy Clin Immunol. 2022 Jan;149(1):445-450. doi: 10.1016/j.jaci.2021.06.007. Epub 2021 Jun 16.

Abstract

Background: Identification of biomarkers associated with immune-mediated diseases in 22q11.2 deletion syndrome is an evolving field.

Objectives: We sought to use a carefully phenotyped cohort to study immune parameters associated with autoimmunity and atopy in 22q11.2 deletion syndrome to define biomarkers associated with immune-mediated disease in this syndrome.

Methods: Chart review validated autoimmune disease and atopic condition diagnoses. Laboratory data were extracted for each subcohort and plotted according to age. A random-effects model was used to define statistical significance.

Results: CD19, CD4, and CD4/45RA lymphocyte populations were not different from the general cohort for patients with atopic conditions. CD4/45RA T cells were significantly lower in the subjects with immune thrombocytopenia compared with the general cohort, and CD4 T-cell counts were lower in patients with autoimmune thyroid disease.

Conclusions: The mechanisms of autoimmunity in cytopenias may be distinct from those of solid-organ autoimmunity in 22q11.2 deletion syndrome. This study identifies potential biomarkers for risk stratification among commonly obtained laboratory studies.

Keywords: DiGeorge; atopy; autoimmunity; dysregulation; homeostatic proliferation.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Adolescent
  • Adult
  • Autoimmune Diseases / immunology*
  • CD4 Lymphocyte Count
  • CD4-Positive T-Lymphocytes / immunology*
  • Child
  • Child, Preschool
  • DiGeorge Syndrome / immunology*
  • Female
  • Humans
  • Hypersensitivity, Immediate / immunology*
  • Infant
  • Male
  • Phenotype
  • Young Adult