Severe hypoglycemia secondary to methimazole-induced insulin autoimmune syndrome in a 16 year old African-American male: Hypoglycemia due to insulin autoimmune syndrome
Open accessAbstract
Insulin autoimmune syndrome (IAS) or Hirata's disease is a rare disorder characterized by hypoglycemia secondary to insulin autoantibodies (IAb). Over 200 patients have been described from Japan with significantly less numbers being reported from outside the Orient. IAS is more common in patients older than 40 yr of age with reports in the pediatric age group being notably rarer. Exposure to sulfhydryl group containing medications is implicated in the pathogenesis of this syndrome. In this report, we describe a case of IAS in an African-American adolescent. A 16-yr-old healthy African-American male was diagnosed with Graves' disease and started on Methimazole. Four weeks later, he was found unconscious and hypoglycemic (blood sugar 1.5 mmol/L). Evaluation was negative for insulinoma. Insulin antibodies were positive. Oral glucose tolerance test revealed elevated free insulin concentrations with disproportionately elevated total insulin levels. The patient was started on prednisone, diazoxide, and propranolol for management of IAS and hyperthyroidism. Thyroid radio-ablation was subsequently undertaken. The doses of prednisone and diazoxide were tapered and these medications discontinued after 9 months. The insulin antibody levels decreased gradually and became undetectable in 6 months with resolution of the hypoglycemia.
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Rare cases of insulin autoimmune syndrome with symptomatic hypoglycemia have been reported in patients treated with MMI.
“Rare cases of insulin autoimmune syndrome with symptomatic hypoglycemia have been reported in patients treated with MMI (146,147).”
From 2016 American Thyroid Association Guidelines for Diagnosis and Management of Hyperthyroidism and Other Causes of Thyrotoxicosis (2016) · cites this paper as [146]
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“Rare cases of insulin autoimmune syndrome with symptomatic hypoglycemia have been reported in patients treated with MMI (146,147).”
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Background
- Authors
- Gomez Cruz, M. J., Jabbar, M., Saini, N., Eng, D., Crawford, B., Vazquez, D. M., Menon, R., & Chen, M.
- Affiliations
- Department of Pediatrics and Communicable Diseases; C.S. Mott Children's Hospital and the University of Michigan; Ann Arbor; MI; USA
- Hurley Medical Center (US)
- Geisinger Medical Center (US)
- University of Michigan (US)
- Corresponding author
- Chen, Ming
- ORCID
- Chen, Ming
- Venue
- Pediatric Diabetes
- Publisher
- Hindawi Limited
- Published
- 2012-07-03
- Volume
- 13
- Issue
- 8
- Pages
- 652-655
- ISSN
- 1399-543X (ISSN-L)
- OpenAlex
- W1910418453
- Cited by
- 34
- References
- 16
- Citations by year
- 2026: 1 · 2025: 2 · 2024: 2 · 2023: 2 · 2022: 1 · 2021: 1 · 2020: 3 · 2019: 2 · 2018: 5 · 2017: 2 · 2016: 5 · 2015: 3 · 2014: 2 · 2013: 3
- FWCI
- 1.53
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Pediatric Diabetes (publishedVersion)
- PubMed (publishedVersion)
- Deep Blue (University of Michigan) (submittedVersion)
- Funders
- University of Michigan
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Diabetes and associated disorders
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Diabetes Management and Research
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Adrenal Hormones and Disorders
- Keywords
- insulin autoimmune syndrome, hypoglycemia, methimazole, insulin autoantibodies, Graves' disease, hyperthyroidism, prednisone, diazoxide, propranolol, adolescents, African Americans
- MeSH terms
- Adolescent, Autoantibodies, Autoimmune Diseases, Black or African American, Diazoxide, Graves Disease, Humans, Hypoglycemia, Insulin, Male, Methimazole, Prednisone, Propranolol, Antithyroid Agents
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