Insulin autoimmune syndrome in an Argentine woman taking α-lipoic acid: A case report and review of the literature. (December 2018)

• Record Type: Journal Article
• Authors: Izzo, Valentina; Greco, Carla; Corradini, Diana; Infante, Marco; Staltari, Maria Teresa; Romano, Maria; Bellia, Alfonso; Lauro, Davide; Uccioli, Luigi
• Is Part Of: SAGE open medical case reports. Volume 6(2018)
• Publication Date: 2018-12
• Subjects: Insulin autoimmune syndrome -- alfa-lipoic acid -- hypoglycaemia -- Hirata disease -- antibodies; Clinical medicine -- Case studies -- Periodicals; 616.09
• DOI: 10.1177/2050313X18819601
• Languages: English
• ISSNs: 2050-313X

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.