title: Severe hypoglycemia secondary to methimazole-induced insulin autoimmune syndrome in a 16 year old African-American male.
abstract: 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.
authors: Department of Pediatrics and Communicable Diseases, C.S. Mott Children's Hospital and the University of Michigan, Ann Arbor, MI, USA.
authors: Michelle J
authors: MJ
authors: Gomez Cruz
authors: Muhammad
authors: M
authors: Jabbar
authors: Natinder
authors: N
authors: Saini
authors: Donna
authors: D
authors: Eng
authors: Brandon
authors: B
authors: Crawford
authors: Delia M
authors: DM
authors: Vazquez
authors: Ram
authors: R
authors: Menon
authors: Ming
authors: M
authors: Chen
mesh_terms: D000293:Adolescent; D001741:Black or African American; D013956:Antithyroid Agents / Q000009:adverse effects*; D001323:Autoantibodies / Q000097:blood; D001327:Autoimmune Diseases / Q000139:chemically induced* / Q000150:complications / Q000188:drug therapy; D003981:Diazoxide / Q000627:therapeutic use; D006111:Graves Disease / Q000188:drug therapy / Q000532:radiotherapy; D006801:Humans; D007003:Hypoglycemia / Q000276:immunology*; D007328:Insulin / Q000276:immunology*; D008297:Male; D008713:Methimazole / Q000009:adverse effects*; D011241:Prednisone / Q000627:therapeutic use; D011433:Propranolol / Q000627:therapeutic use
chemical_list: D013956:Antithyroid Agents; D001323:Autoantibodies; D007328:Insulin; D008713:Methimazole; D011433:Propranolol; D003981:Diazoxide; D011241:Prednisone