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SAGE Open Med Case Rep. 2018 Dec 26;6:2050313X18819601. doi: 10.1177/2050313X18819601. eCollection 2018.

Insulin autoimmune syndrome in an Argentine woman taking α-lipoic acid: A case report and review of the literature.

SAGE open medical case reports

Valentina Izzo, Carla Greco, Diana Corradini, Marco Infante, Maria Teresa Staltari, Maria Romano, Alfonso Bellia, Davide Lauro, Luigi Uccioli

Affiliations

  1. Department of Systems Medicine, University of Rome Tor Vergata, Rome, Italy.

PMID: 30627435 PMCID: PMC6311546 DOI: 10.1177/2050313X18819601

Abstract

Insulin autoimmune syndrome is an unusual cause of spontaneous hypoglycaemia in non-Asian populations. In the majority of cases, this syndrome appears a few weeks after the administration of drugs containing a sulfhydryl group. A strong association between this syndrome and HLA-DR4 has been shown. Only seven cases have been described in non-Asian patients. We report the first case of insulin autoimmune syndrome in an Argentine woman taking alfa-lipoic acid. She developed hypoglycaemic symptoms approximately 1 month after starting therapy. Blood sampling collected during an episode of symptomatic hypoglycaemia showed low blood glucose level (2.39 mmol/L), high level of serum insulin (1971.55 pmol/L), inappropriately high level of C-peptide (2.36 nmol/L) and high levels of insulin antibodies (274.78 IU/mL). HLA-DNA typing identified DRB1*04:03. Due to the widespread use of alfa-lipoic acid for its antioxidant properties, clinicians should be aware that it may trigger an autoimmune hypoglycaemia in people with a genetic predisposition.

Keywords: Hirata disease; Insulin autoimmune syndrome; alfa-lipoic acid; antibodies; hypoglycaemia

Conflict of interest statement

Declaration of conflicting interests: The author(s) declare no potential conflict of interest with respect to the research, authorship and publication of the article.

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