ArticleJournal of clinical immunology2013
Anti-cytokine autoantibodies preceding onset of autoimmune polyendocrine syndrome type I features in early childhood.
Article in Journal of clinical immunology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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Who cites it
33 citing papers in PubMed, 81 citations in OpenAlex.
- Multiplex Autoantibody Detection in Patients with Autoimmune Polyglandular Syndromes.International journal of molecular sciences · 2021Trial
- Clinical, genetic, and immunologic features of APS-1 patients from the Middle East, and a review of the literature.Journal of human immunity · 2026Article
- Rare pediatric Saccharomyces cerevisiae fungemia with neutralizing anti-IL-23 autoantibodies: a case report.BMC pediatrics · 2026Article
- Factors associated with and kinetics of anti-IFN-α autoantibodies inThe journal of allergy and clinical immunology. Global · 2025Article
- Human immunity to fungal infections.The Journal of experimental medicine · 2025Review
- Thymus research in relation to myasthenia gravis: a new perspective on cell subpopulations and future directions.Frontiers in immunology · 2025Review
- Anti-cytokine autoantibodies: mechanistic insights and disease associations.Nature reviews. Immunology · 2024Review
- Novel Insights into the Autoimmunity from the Genetic Approach of the Human Disease.Advances in experimental medicine and biology · 2024Article
- Interferon autoantibodies as signals of a sick thymus.Frontiers in immunology · 2024Review
- Autoantibodies to Interferons in Infectious Diseases.Viruses · 2023Review
- Human autoantibodies underlying infectious diseases.The Journal of experimental medicine · 2022Article
- Persistently Increased Anti-cytokine Antibodies Without Clinical Disease in a Boy with APS1 Genotype.Journal of clinical immunology · 2022Article
- B Cells and Autoantibodies in AIRE Deficiency.Biomedicines · 2021Review
- Infection-induced inflammation from specific inborn errors of immunity to COVID-19.The FEBS journal · 2021Review
- Thymus and autoimmunity.Seminars in immunopathology · 2021Review
- Article
- AIRE expression controls the peripheral selection of autoreactive B cells.Science immunology · 2019Observational
- Lessons from primary immunodeficiencies: Autoimmune regulator and autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy.Immunological reviews · 2019Review
- Oral microbiota in autoimmune polyendocrine syndrome type 1.Journal of oral microbiology · 2018Article
- Autoimmune polyendocrine syndrome type 1 in an Indian cohort: a longitudinal study.Endocrine connections · 2017Article
Corrections and comments
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Authors and funding
16 authors at 10 institutions in 8 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
purposeAlmost all patients with autoimmune polyendocrine syndrome (APS)-I have high titer neutralizing autoantibodies to type I interferons (IFN), especially IFN-ω and IFN-α2, whatever their clinical features and onset-ages. About 90 % also have antibodies to interleukin (IL)-17A, IL-17F and/or IL-22; they correlate with the chronic mucocutaneous candidiasis (CMC) that affects ~90 % of patients. Our aim was to explore how early the manifestations and endocrine and cytokine autoantibodies appear in young APS-I patients. That may hold clues to very early events in the autoimmunization process in these patients.
methodsClinical investigations and autoantibody measurements in 13 APS-I patients sampled before age 7 years, and 3 pre-symptomatic siblings with AIRE-mutations in both alleles.
resultsAntibody titers were already high against IFN-α2 and IFN-ω at age 6 months in one sibling-8 months before onset of APS-I-and also against IL-22 at 7 months in another (still unaffected at age 5 years). In 12 of the 13 APS-I patients, antibody levels were high against IFN-ω and/or IL-22 when first tested, but only modestly positive against IFN-ω in one patient who had only hypo-parathyroidism. Endocrine organ-specific antibodies were present at age 6 months in one sibling, and as early as 36 and 48 months in two of the six informative subjects.
conclusionThis is the first study to collate the onset of clinical features, cytokine and endocrine autoantibodies in APS-I infants and siblings. The highly restricted early autoantibody responses and clinical features they show are not easily explained by mere loss of broad-specific self-tolerance inducing mechanisms, but hint at some more sharply focused early event(s) in autoimmunization.
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