Authors
Sai Prasad, Anukriti Sharma, Vidhi Parmar, Tanisha Jindal, Darpan Kothia, Deep Bavaria
Published in
JCEM case reports. Volume 4. Issue 8. Pages luag190. Epub Jul 13, 2026.
Abstract
Insulin autoimmune syndrome (IAS), also known as Hirata disease, is a rare cause of spontaneous hypoglycemia due to circulating insulin autoantibodies, most commonly described in East Asian individuals and those exposed to sulfhydryl-containing drugs. We describe a 58-year-old South Asian man with type 2 diabetes mellitus who presented with recurrent fasting and postprandial hypoglycemia despite cessation of all glucose-lowering medications. A spontaneous episode confirmed Whipple triad: plasma glucose 38 mg/dL (SI: 2.1 mmol/L; reference range, 70-99 mg/dL [SI: 3.9-5.5 mmol/L]), markedly elevated serum insulin at 156 µIU/mL (SI: 1083 pmol/L; reference range, 2.6-24.9 µIU/mL [SI: 18-173 pmol/L]), and C-peptide of 5.8 ng/mL (SI: 1.9 nmol/L; reference range, 0.5-2.0 ng/mL [SI: 0.17-0.66 nmol/L]), with a negative sulfonylurea screen and markedly elevated insulin autoantibody titers. Polyethylene glycol precipitation confirmed antibody-bound insulin sequestration. Workup identified IgG-kappa monoclonal gammopathy of undetermined significance as the probable precipitant. Dietary modification, oral glucocorticoids, and acarbose achieved near-complete resolution of hypoglycemia with declining autoantibody titers over 4 weeks. This case highlights the importance of considering IAS in unexplained hyperinsulinemic hypoglycemia and the need to screen for plasma cell dyscrasias in atypical presentations.
PMID:
42438805
Bibliographic data and abstract were imported from PubMed on 13 Jul 2026.
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