Noninfectious Complications in B-Lymphopenic Common Variable Immunodeficiency

J Investig Allergol Clin Immunol. 2024 Jul;34(4):233-245. doi: 10.18176/jiaci.0902.

Abstract

Background: Common variable immunodeficiency (CVID) is considered the most symptomatic type of inborn errors of immunity in humans. Along with infectious complications, which have numerous consequences, noninfectious complications are a major challenge among CVID patients.

Methods: All CVID patients registered in the national database were included in this retrospective cohort study. Patients were divided into 2 groups based on the presence of B-cell lymphopenia. Demographic characteristics, laboratory findings, noninfectious organ involvement, autoimmunity, and lymphoproliferative diseases were evaluated.

Results: Among 387 enrolled patients, 66.4% were diagnosed with noninfectious complications and 33.6% with isolated infectious presentations. Enteropathy, autoimmunity, and lymphoproliferative disorders were reported in 35.1%, 24.3%, and 21.4% of patients, respectively. Some complications, including autoimmunity and hepatosplenomegaly, were reported to be significantly more frequent among patients with B-cell lymphopenia. As for organ involvement, the dermatologic, endocrine, and musculoskeletal systems were predominantly affected in CVID patients with B-cell lymphopenia. Among autoimmune manifestations, the frequency of rheumatologic, hematologic, and gastrointestinal autoimmunity was reported to be higher than that of other types of autoimmunity not associated with B cell-lymphopenia. Furthermore, hematological cancers, particularly lymphoma, were the most common type of malignancy. The mortality rate was 24.5%, and respiratory failure and malignancies were the most common causes of death, with no significant differences between the 2 groups.

Conclusions: Considering that some of the noninfectious complications might be associated with B-cell lymphopenia, regular patient monitoring and follow-up with proper medication (in addition to immunoglobulin replacement therapy) are highly recommended to prevent sequelae and increase patient quality of life.

Keywords: Autoimmunity; Common variable immunodeficiency; Immune dysregulation; Inborn errors of immunity; Malignancy; Primary immunodeficiency.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Autoimmunity
  • B-Lymphocytes* / immunology
  • Child
  • Common Variable Immunodeficiency* / complications
  • Common Variable Immunodeficiency* / immunology
  • Female
  • Humans
  • Lymphopenia* / immunology
  • Male
  • Middle Aged
  • Retrospective Studies
  • Young Adult