Abstract
A 3-yr-old female patient exhibited interleukin 12 (IL-12) deficiency that was associated with recurrent episodes of pneumococcal pneumonia with sepsis and other infections in the absence of fevers. The patient's peripheral blood mononuclear cells (PBMCs) exhibited normal proliferative responses to antigens. Immune responses, including in vivo production of antibodies to diphtheria, tetanus, or pneumococcal antigens, were normal. Ig levels and B cell and T cell phenotypes were also normal. In contrast, IL-12 p70 heterodimer production was undetectable by using supernatants of the patient's stimulated PBMCs when compared with control cells treated similarly. Although present, interferon gamma (IFN-gamma) was reduced. The addition of recombinant IFN-gamma to control cells enhanced the production of IL-12 by up to sixfold. By contrast, IL-12 was undetectable in supernatants of the patient's cells in the presence of recombinant IFN-gamma. IL-12 p40 subunit mRNA by using the patient's PBMCs after stimulation with Staphylococcus aureus Cowan strain 1 or lipopolysaccharide was also undetectable by reverse transcription-PCR when compared with control cells. Production of IL-2, IL-6, tumor necrosis factor alpha, or IFN-gamma of the patient's PBMCs after appropriate stimulation was observed. This patient has either a defect in Staphylococcus aureus Cowan strain 1-lipopolysaccharide- or staphylococcal enterotoxin A-induced signaling pathways for the activation of IL-12 p40 gene expression, or an abnormality in the IL-12 p40 gene itself.
MeSH Terms
Adolescent
Antigens, CD/analysis
Cells, Cultured
Child, Preschool
Communicable Diseases/genetics,immunology
Consanguinity
Female
Humans
Immunologic Deficiency Syndromes/complications,genetics,immunology
Immunophenotyping
Incest
Interferon-gamma/analysis
Interleukin-12/analysis,deficiency,genetics
Interleukin-2/analysis
Lymphocyte Activation
Lymphocytes/classification,immunology
Polymerase Chain Reaction
Recurrence
Reference Values
Tumor Necrosis Factor-alpha/analysis
Chemicals
Antigens, CD
Interleukin-2
Tumor Necrosis Factor-alpha
Interleukin-12
Interferon-gamma
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Haraguchi S
Department of Pediatrics, University of South Florida College of Medicine/All Children's Hospital, 801 Sixth Street South, St. Petersburg, FL 33701, USA.
Day N K
Nelson R P
Emmanuel P
Duplantier J E
Christodoulou C S
Good R A
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