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[Autoimmune polyendocrinopathy with IGA deficiency]

U Schwarz, B Lämmle, P Six

    Schweizerische Medizinische Wochenschrift
    |December 2, 1978
    PubMed
    Summary
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    This case report details a patient with Addisonian crisis, revealing autoimmune polyendocrinopathy and diminished immunoglobulin A (IgA) levels. Prolonged diphenylhydantoin use may link IgA deficiency with this pluriglandular failure.

    Area of Science:

    • Endocrinology
    • Immunology
    • Pharmacology

    Context:

    • Presents a rare case of a 35-year-old female experiencing Addisonian crisis.
    • Highlights the detection of primary adrenal, ovarian, and thyroid failure.
    • Notes the presence of autoantibodies against adrenal and thyroid tissues.

    Purpose:

    • To report a unique case of IgA deficiency associated with autoimmune polyendocrinopathy.
    • To explore the potential link between prolonged diphenylhydantoin therapy and immunological disturbances.
    • To discuss the coincidence of IgA deficiency and autoimmune diseases.

    Summary:

    • The patient presented with Addisonian crisis, subsequently diagnosed with autoimmune polyendocrinopathy (adrenal, ovarian, thyroid failure).
    • Diminished serum IgA levels were observed, alongside positive autoantibodies.

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  • A history of 20-year diphenylhydantoin treatment was noted, a drug known to cause immunological issues including IgA depression.

    • Impact:

    • This case may be the first documented instance of IgA deficiency linked to autoimmune polyendocrinopathy.
    • Suggests a potential etiological role for long-term diphenylhydantoin use in developing both IgA deficiency and pluriglandular autoimmune failure.
    • Underscores the known association between IgA deficiency and autoimmune conditions.