Hypoglycemic disorders are rare in persons without diabetes, and clinical evaluation to identify its etiology can be challenging. We present a case of insulin autoimmune syndrome induced by carbimazole in a middle-aged Chinese man with underlying Graves’ disease, which was managed conservatively with a combination of dietary modification and alpha-glucosidase inhibitor.
Hypoglycemia ; Hyperinsulinism ; Insulin Antibodies

Herein, we report a case of unusually elevated serum insulin level as a result of increased anti-insulin antibody (IA)-bound insulin after continuous subcutaneous insulin infusion therapy. Detecting free insulin (unbound IAs) levels after polyethylene glycol pre-treatment could be useful to assess functional insulin levels in diabetic patients receiving insulin therapy. The E170 insulin assay can estimate total insulin (bound IAs and free insulin) levels, but it does not measure the levels of exogenous insulin analogues.
Humans ; Insulin ; Insulin Antibodies ; Polyethylene Glycols

Hypoglycemia in diabetic patients is usually caused by excessive exogenous insulin or the administration of an insulin secretagogue relative to the prevailing glucose concentration. Thus, the clinical manifestations of hypoglycemia are usually not observed in diabetic patients after either insulin or an oral hypoglycemic agent is discontinued. In contrast, diabetic ketoacidosis results from relative or absolute insulin deficiency. Although about 40% of diabetic patients who inject human insulin have insulin antibodies, these antibodies seldom significantly affect the glycemic control. It has not been reported in the literature that insulin antibody in the setting of human insulin therapy is associated with diabetic ketoacidosis and subsequent hypoglycemia. We describe here a rare case of spontaneous hypoglycemia due to insulin antibody after the improvement of diabetic ketoacidosis in a patient with type 2 diabetes mellitus and who had been treated with human insulin.
Antibodies ; Diabetes Mellitus ; Diabetes Mellitus, Type 2 ; Diabetic Ketoacidosis ; Glucose ; Humans ; Hypoglycemia ; Insulin ; Insulin Antibodies

PURPOSE: The purpose of this study was to determine whether there is any difference in the clinical and laboratory characteristics of patients with autoantibody-positive and patients with autoantibody-negative type 1 diabetes at initial presentation. METHODS: We analyzed 96 patients under 18 years of age with newly diagnosed type 1 diabetes. One or both of the pancreatic autoantibodies-glutamic acid decarboxylase autoantibodies (GADA) and insulin autoantibody (IAA)-were measured in all patients, and we reviewed clinical and laboratory characteristics according to the presence of these autoantibodies. RESULTS: GADA was examined in 48 of 87 patients, and 55.2% of patients were positive. IAA was checked in 88 patients, and 39.8% were positive. Both GADA and IAA were measured in 83 patients, and 22.8% had both antibodies. The patients who had one or both autoantibodies (autoantibody-positive group) were younger than those not having any autoantibody (autoantibody-negative group). The autoantibody-positive group had lower BMI, corrected sodium level, and serum effective osmolarity, compared to the autoantibody-negative group (P<0.05). Similar differences were found between the GADA-positive and GADA-negative groups. However, there were no significant differences between the IAA-positive and IAA-negative groups. CONCLUSION: The prevalence of pancreatic autoantibodies was significantly higher in the under-6 years age group than in the other age groups. These findings suggest that measurement of autoantibodies at the initial diagnosis of diabetes is very useful for detecting immune-mediated type 1 diabetes and providing intensive insulin therapy, especially in younger children.
Antibodies ; Autoantibodies ; Child ; Humans ; Insulin ; Osmolar Concentration ; Prevalence ; Sodium

Hyperinsulinemic hypoglycemia in the absence of exogenous insulin use is caused by disorders such as insulinoma, diffuse beta-cell hyperplasia/nesidioblastosis, and autoimmune hypoglycemia. Nesidioblastosis is a rare cause of hypoglycemia in adults, accounting for 0.5~7.0% of organic hyperinsulinemia cases. Although pancreatic resection is considered the best treatment modality for curing nesidioblastosis, there is no consensus regarding the indications for and extent of the surgery due to its high risk and complication rate. A 75-year-old woman presented with an altered mental state, a mass suspected of being an insulinoma, and insulin receptor antibodies. The patient underwent surgery because of recurrent life-threatening hypoglycemia. Postoperative pathology of her pancreas revealed nesidioblastosis.
Accounting ; Adult ; Aged ; Antibodies ; Consensus ; Female ; Humans ; Hyperinsulinism ; Hypoglycemia ; Insulin ; Insulinoma ; Nesidioblastosis ; Pancreas ; Receptor, Insulin

Autoimmune insulin syndrome is characterized by a combination of fasting hypoglycemia, high total serum insulin concentrations, and the presence of autoantibodies to the native human insulin in serum. A healthy 69-year-old man developed spontaneous hypoglycemia after being given some medications for the common cold. His laboratory finding shows endogenous hyperinsulinism. Therefore, an insulinoma was suspected but the imaging study shows no mass lesion in the pancreas and the anti-insulin antibody titer was high(>100U/mL). He had no specific medication history except for a treatment of a current upper respiratory infection (URI). Among the URI medication, it was found that N-acetylcysteine contains a sulfhydryl group and could induce autoimmunization. The patient was treated with corticosteroid for 3 months. His anti-insulin antibody titer had decreased to normal level and has not shown any hypoglycemic episodes for more than one and a half years. We present this case with a review of relevant literature.
Acetylcysteine ; Aged ; Autoantibodies ; Common Cold ; Humans ; Hyperinsulinism ; Hypoglycemia ; Insulin Antibodies ; Insulin* ; Insulinoma ; Pancreas

Hypoglycemia was detected in a 15-year-old girl due to loss of consciousness. She was diagnosed with Graves' disease and was being treated with methimazole for the past 4 months. A paradoxically increased insulin levels was found when she suffered from the hypoglycemic episode. An imaging study showed no mass lesion in the pancreas, and insulin antibodies were found in the serum. She was diagnosed with insulin autoimmune syndrome. Her HLA typing was performed, and it revealed HLA-DRB1 *04:06. The patient was treated with a corticosteroid for 2 months. After discontinuing the steroid, the insulin antibody titer decreased dramatically, and she did not have any episode of hypoglycemia since. This is the first report of insulin autoimmune syndrome in a Korean girl, and we have revealed the connection between HLA type and insulin autoimmune syndrome in Korea.
Adolescent ; Graves Disease ; Histocompatibility Testing ; HLA-DRB1 Chains ; Humans ; Hypoglycemia ; Insulin ; Insulin Antibodies ; Korea ; Methimazole ; Pancreas ; Unconsciousness

Insulin autoimmune syndrome (IAS) is characterized by spontaneous hypoglycemia, extremely high serum insulin levels, and high titers of autoantibodies against endogenous insulin, in the absence of exogenous insulin injection. IAS often occurs following exposure to sulfhydryl-containing drugs, including alpha-lipoic acid (ALA). A 30-year-old woman without diabetes visited our outpatient clinic with recurrent hypoglycemia. She had been taken ALA for weight reduction since 3 weeks ago. Further hypoglycemia work up revealed very high insulin levels, C-Peptide levels and positive insulin antibodies. And conventional imaging examinations were negative for insulinoma or other pancreatic tumors. Finally, the diagnosis of Insulin autoimmune syndrome (IAS) was made. Following the cessation of ALA, hypoglycemia improved, with no medication, and the patient experienced no further hypoglycemic attacks over the next month. The use of ALA as a nutritional supplement is increasing. We report a case of IAS associated with ALA in a non-diabetic patient.
Adult ; Ambulatory Care Facilities ; Autoantibodies ; C-Peptide ; Diagnosis ; Female ; Humans ; Hypoglycemia ; Insulin Antibodies ; Insulin* ; Insulinoma ; Thioctic Acid* ; Weight Loss

BACKGROUND: Endogenous hyperinsulinemic hypoglycemia (EHH) is characterized by an inappropriately high plasma insulin level, despite a low plasma glucose level. Most of the EHH cases are caused by insulinoma, whereas nesidioblastosis and insulin autoimmune syndrome (IAS) are relatively rare. METHODS: To evaluate the relative frequencies of various causes of EHH in Korea, we retrospectively analyzed 84 patients who were diagnosed with EHH from 1998 to 2012 in a university hospital. RESULTS: Among the 84 EHH patients, 74 patients (88%), five (6%), and five (6%) were diagnosed with insulinoma, nesidioblastosis or IAS, respectively. The most common clinical manifestation of EHH was neuroglycopenic symptoms. Symptom duration before diagnosis was 14.5 months (range, 1 to 120 months) for insulinoma, 1.0 months (range, 6 days to 7 months) for nesidioblastosis, and 2.0 months (range, 1 to 12 months) for IAS. One patient, who was diagnosed with nesidioblastosis in 2006, underwent distal pancreatectomy but was later determined to be positive for insulin autoantibodies. Except for one patient who was diagnosed in 2007, the remaining three patients with nesidioblastosis demonstrated severe hyperinsulinemia (157 to 2,719 microIU/mL), which suggests that these patients might have had IAS, rather than nesidioblastosis. CONCLUSION: The results of this study suggest that the prevalence of IAS may be higher in Korea than previously thought. Therefore, measurement of insulin autoantibody levels is warranted for EHH patients, especially in patients with very high plasma insulin levels.
Autoantibodies ; Autoimmune Diseases ; Blood Glucose ; Diagnosis ; Humans ; Hyperinsulinism ; Hypoglycemia* ; Insulin ; Insulin Antibodies ; Insulinoma ; Korea ; Nesidioblastosis ; Pancreatectomy ; Plasma ; Prevalence ; Retrospective Studies

Insulin autoimmune syndrome (IAS) is characterized by spontaneous hypoglycemia, extremely high serum insulin levels, and high titers of autoantibodies against endogenous insulin, in the absence of exogenous insulin injection. IAS often occurs following exposure to sulfhydryl-containing drugs, including alpha-lipoic acid (ALA). A 30-year-old woman without diabetes visited our outpatient clinic with recurrent hypoglycemia. She had been taken ALA for weight reduction since 3 weeks ago. Further hypoglycemia work up revealed very high insulin levels, C-Peptide levels and positive insulin antibodies. And conventional imaging examinations were negative for insulinoma or other pancreatic tumors. Finally, the diagnosis of Insulin autoimmune syndrome (IAS) was made. Following the cessation of ALA, hypoglycemia improved, with no medication, and the patient experienced no further hypoglycemic attacks over the next month. The use of ALA as a nutritional supplement is increasing. We report a case of IAS associated with ALA in a non-diabetic patient.
Adult ; Ambulatory Care Facilities ; Autoantibodies ; C-Peptide ; Diagnosis ; Female ; Humans ; Hypoglycemia ; Insulin Antibodies ; Insulin ; Insulinoma ; Thioctic Acid ; Weight Loss