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We report on two cases of type C insulin resistance syndrome(TCIRS) admitted to the Department of Endocrinology, Peking Union Medical College Hospital from January 2000 to December 2020. Both patients presented with persistent hyperglycemia, low immunoreactive insulin, extreme insulin resistance, high insulin autoantibodies, high total insulin, and large insulin antibody pool. TCIRS is marked by extreme insulin resistance with ketoacidosis and respond to medium to high doses glucocorticoids rather than plasmapheresis.
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Objective:A retrospective study was conducted to analysis the clinical characteristics of 7insulin autoimmune syndrome (IAS) patients.Methods:Clinical data were collected by searching the computerized database.Results:The male-female ratio of these seven patients was 4:3; age of the four patients was between 60-70 years old;two patients with the history of Hashimoto's disease. Of the seven cases, six wereexogenous IAS. The level of insulin was excessively high, the level of C-peptide was not low, and insulin auto-antibodies (IAA) were positive of all the seven patients. The lowest blood glucose of one patient was 4.2 mmol/L. The insulin to C-peptide molar ratios were >1 in five patients. Symptoms were relieved after discontinuing use of the suspicious drugs, small frequent meals, taking acarbose and metformin.Conclusions:IAS should not be easily excluded in patients without hypoglycemia record. For diabetes patients receiving insulin therapy, exogenous IAS might be mistaken as hypoglycemia induced by insulin overdose. The identification of the genotype might be meaningful in the diagnosing and prevention of IAS.
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AIM: To explore the clinical features of insulin autoimmune syndrome (IAS) induced by methimazole in Chinese population. METHODS: The literature on IAS cases caused by methimazole in the Chinese population published before and after June 30, 2019 was collected for retrospective analysis.RESULTS:The age of onset of men was earlier than that of women, and the sex ratio was 1:2.31 in 95 patients.After taking methimazole 30 mg/d, IAS occurred most from one month to three months,characterized by neuropathic hypoglycemia as the first symptoms at night and early in the morning,blood sugar below 2 mmol/L,insulin concentration≥100 mU/L, IAA positive and no obvious abnormalities in pancreas imaging.The symptoms gradually relieved after symptomatic treatment and stopped taking methimazole. There was no significant difference in the time of hypoglycemia disappearance between the 54 patients who received hormone therapy and non-hormone therapy.CONCLUSION:Methimazole-induced IAS is a clinically rare autoimmune disease. It should be treated promptly occuring hypoglycemia or hyperglycemia during medication.The prognosis of IAS is generally good after proper treatment.
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A 52-year-old woman was transported for reduced consciousness. Her blood glucose was only 19 mg/dL, but her blood immunoreactive insulin and insulin antibody levels were high at 250 μU/mL and 50 U/mL, respectively. She had no history of insulin treatment, but she had been taking coenzyme Q10 supplements for three months. Her human leukocyte antigen serotype was DR4. After stopping coenzyme Q10, her hypoglycemia disappeared and immunoreactive insulin and insulin antibody levels normalized. Based on the above, she was diagnosed with insulin autoimmune syndrome caused by coenzyme Q10. It is necessary to be aware of the onset of insulin autoimmune syndrome due to coenzyme Q10. Its pathogenesis requires clarification.
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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.
Subject(s)
Adult , Female , Humans , Ambulatory Care Facilities , Autoantibodies , C-Peptide , Diagnosis , Hypoglycemia , Insulin Antibodies , Insulin , Insulinoma , Thioctic Acid , Weight LossABSTRACT
Insulin autoimmune syndrome ( IAS) is a rare type of hypoglycemia. Chinese herbal extract may induce IAS, which seems to be associated with some Chinese herb medicine ingredients of paste which contains sulfhydryl group. In order to attract the attention of the clinicians, we should raise awareness of the disease, and avoid unnecessary operation or serious adverse consequences. This paper reviewed domestic reports in lately 30 years of diagnosis and treatment of IAS, evaluated traditional Chinese medicine and traditional Chinese medicine compound preparations which contain sulfhydlyl group.
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A 65-year-old male presented with recurrent palpitation and fatigue over one year. Lab tests revealed him with hyperinsulinaemic hypoglycemia. Insulin autoimmune antibody was repeatedly negative. Imaging of the pancreas seemed to be normal. Insulin-insulin autoimmune antibody complexes were detected by polyethylene glycol precipitation and gel filtration chromatography, thus the diagnosis of insulin autoimmune syndrome was suggested. By adjusting diet and administration of acarbose, all the symptoms were evidently relieved.
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A 65-year-old male presented with recurrent palpitation and fatigue over one year. Lab tests revealed him with hyperinsulinaemic hypoglycemia. Insulin autoimmune antibody was repeatedly negative. Imaging of the pancreas seemed to be normal. Insulin-insulin autoimmune antibody complexes were detected by polyethylene glycol precipitation and gel filtration chromatography, thus the diagnosis of insulin autoimmune syndrome was suggested. By adjusting diet and administration of acarbose, all the symptoms were evidently relieved.
ABSTRACT
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.
Subject(s)
Adult , Female , Humans , Ambulatory Care Facilities , Autoantibodies , C-Peptide , Diagnosis , Hypoglycemia , Insulin Antibodies , Insulin , Insulinoma , Thioctic Acid , Weight LossABSTRACT
Objective:To know the clinical features of insulin autoimmune syndrome to improve diagnostic level. Methods:Focused oninsulin autoimmune syndrome ( IAS ), to make retrospective analysis by summarizing medical history, clinical manifestations,insulin levels,treatment and prognosis of 71 IAS cases recently fifteen years reported in the literature on CNKI. Results:Among the 71 IAS cases,there were 40 cases with a history of hyperthyroidism,31 cases taking methimazole before onset and 43 cases with hypoglycemic episodes with clear regularity, of which 30 cases hypoglycemic episodes occurred during the night or on early morning. 35 cases had disorders of consciousness and such patients had lower blood glucose level and higher insulin level; 36 cases received hormone therapy with shorter time for IAA turning negative,and 26 cases only stopped taking the causative drug;69 cases have a remission,1 case prognosis was unknown due to auto-discharge,and 1 case dies of combined pleural endotheliomas. Conclusion:IAS is one of the important causes of severe spontaneous hypoglycemia and is clinically relatively rare. It often combines with autoimmune diseases,induced by hydroxyl drug and vulnerable to disturbance of consciousness. Patients who have disorders of consciousness should be treated with hormonotherapy in time. Early identification and appropriate treatment generally lead to good prognosis.
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Insulin autoimmune syndrome, a rare cause of endogenous hyperinsulinemic hypoglycemia, is characterized by insulin autoantibody, hyperinsulinemia and fasting hypoglycemia. It is well known that drugs containing a sulfhydryl group such as methimazole or α-mercaptopropionyl glycine can induce insulin autoimmune syndrome. However, insulin autoimmune syndrome caused by anti-tuberculosis treatment is very rare. We report a case of insulin autoimmune syndrome after anti-tuberculosis treatment with a review of the relevant literature.
Subject(s)
Glycine , Hyperinsulinism , Hypoglycemia , Insulin , Methimazole , TuberculosisABSTRACT
Insulin autoimmune syndrome (IAS) is characterized by fasting hypoglycemia, endogenous hyperinsulinemia, and the presence of autoantibodies to insulin or insulin receptor in patients that have never been exposed to exogenous insulin. This syndrome is occasionally accompanied by several autoimmune disorders. There is no reported case of concurrent IAS with Hashimoto's thyroiditis. A 52-year-old female was diagnosed with Hashimoto's thyroiditis and was treated with 25 microg/d levothyroxine for 3 years. Recently, she experienced recurrent fasting hypoglycemic symptoms that disappeared rapidly with a carbohydrate-rich diet, although she had no history of diabetes or insulin use. Blood analysis showed hypoglycemia and elevated serum levels of insulin and C-peptide. Imaging studies did not reveal a mass lesion in the pancreas, and selective calcium-stimulated venous sampling also gave a negative result. However, anti-insulin antibody titer was high and assay for anti-insulin receptor antibody was positive. Here, we report a case of IAS concomitant with Hashimoto's thyroiditis.
Subject(s)
Female , Humans , Middle Aged , Autoantibodies , C-Peptide , Diet , Fasting , Hyperinsulinism , Hypoglycemia , Insulin , Pancreas , Receptor, Insulin , Thyroid Gland , Thyroiditis , ThyroxineABSTRACT
Insulin autoimmune syndrome (IAS) is characterized by spontaneous hypoglycemia caused by insulin autoantibodies in the absence of exogenous insulin administration. Some drugs containing sulfhydryl compounds are known to initiate the onset of IAS. A 67-year-old female who had diabetes for 5 years visited the outpatient clinic at our institution due to diabetic peripheral polyneuropathy. She was prescribed alpha-lipoic acid (ALA), which contains two sulfur atoms. Two weeks later, she complained of recurrent hypoglycemic symptoms. We detected a high level of insulin and high titers of insulin autoantibodies. Her human leukocyte antigen (HLA) genotype included the DRB1*0406 allele, which indicates a high level of susceptibility to IAS. She was treated with prednisolone. After this episode, she experienced two more hypoglycemic events after taking ALA for diabetic neuropathy in other hospitals. As ALA can be used to treat diabetic peripheral polyneuropathy, physician discretion is advised based on the possibility of IAS due to ALA in diabetic patients.
Subject(s)
Aged , Female , Humans , Alleles , Ambulatory Care Facilities , Autoantibodies , Diabetic Neuropathies , Genotype , Hypoglycemia , Insulin Antibodies , Insulin , Leukocytes , Polyneuropathies , Prednisolone , Sulfhydryl Compounds , Sulfur , Thioctic AcidABSTRACT
Insulin autoimmune syndrome (IAS) is characterized by spontaneous hypoglycemia caused by insulin autoantibodies in the absence of exogenous insulin administration. Some drugs containing sulfhydryl compounds are known to initiate the onset of IAS. A 67-year-old female who had diabetes for 5 years visited the outpatient clinic at our institution due to diabetic peripheral polyneuropathy. She was prescribed alpha-lipoic acid (ALA), which contains two sulfur atoms. Two weeks later, she complained of recurrent hypoglycemic symptoms. We detected a high level of insulin and high titers of insulin autoantibodies. Her human leukocyte antigen (HLA) genotype included the DRB1*0406 allele, which indicates a high level of susceptibility to IAS. She was treated with prednisolone. After this episode, she experienced two more hypoglycemic events after taking ALA for diabetic neuropathy in other hospitals. As ALA can be used to treat diabetic peripheral polyneuropathy, physician discretion is advised based on the possibility of IAS due to ALA in diabetic patients.
Subject(s)
Aged , Female , Humans , Alleles , Ambulatory Care Facilities , Autoantibodies , Diabetic Neuropathies , Genotype , Hypoglycemia , Insulin Antibodies , Insulin , Leukocytes , Polyneuropathies , Prednisolone , Sulfhydryl Compounds , Sulfur , Thioctic AcidABSTRACT
We report here the cases of two females with Graves' disease who developed insulin autoimmune syndrome after treatment with methimazole. The patients exhibited a sudden altered mental state after treatment with methimazole for approximately 4 weeks. Patients had hypoglycemia with serum glucose below 70 mg/dL, and laboratory findings showed both high levels of serum insulin and high titers of insulin autoantibodies. The two women had never been exposed to insulin or oral antidiabetic agents, and there was no evidence of insulinoma in imaging studies. After glucose loading, serum glucose, and total insulin levels increased abnormally. One of the patient was found to have HLA-DRB1*0406, which is known to be strongly associated with methimazole-induced insulin autoimmune syndrome. After discontinuation of methimazole, hypoglycemic events disappeared within 1 month. Insulin autoantibody titer and insulin levels decreased within 5 months and there was no further development of hypoglycemic events. We present these cases with a review of the relevant literature.
Subject(s)
Female , Humans , Autoantibodies , Glucose , Graves Disease , HLA-DRB1 Chains , Hypoglycemia , Hypoglycemic Agents , Insulin , Insulinoma , MethimazoleABSTRACT
Objective To analyze the clinical characteristics and prognosis of patients with insulin autoimmune syndrome (IAS) and to get better understanding of IAS by literatures reviewing.Methods Nine cases of IAS who were diagnosed in the General Hospital of the People's Liberation Army from 2001 to 2011 were analyzed.Results All patients had hypoglycemic syndrome and episodes of hypoglycemia during both postprandial and fasting states.Most cases (8/9) were accompanied with autoimmune diseases,including 6 cases of Graves' disease treated with methimazole.The biochemical data showed extremely elevated serum insulin level (9/9) during hypoglycemic episode.For most patients,the tests of insulin autoantibodies were positive (7/9) while results of imaging examinations were negative(8/9).After removal of possible offending medications and with diet treatment,hypoglycemic episodes were ameliorated in 5 of 9 cases.For severe patients,acarbose (1/9) and prednisone (3/9)therapy were useful.During the period of follow-up,four cases experienced no episode of hypoglycemia and 3 cases with markedly reduced episodes.Conclusions IAS is characterized by hypoglycemic episodes,elevated blood insulin levels,and positive insulin autoantibodies.It is strongly related with autoimmune disease and is able to be induced by methimazole.Most patients undergo remission after diet treatment,drug withdrawal,and oral prednisone.
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Insulin autoimmune syndrome (IAS) is rare.A patient with IAS was herewith reported and 50domestic cases reported in the last two decades were reviewed to discuss the approach to this syndrome.In hypoglycemic patients with high insulin level and with Graves' disease and taking tapazole,insulin autoantibody detection is mandatory to avoid misdiagnosis and unnecessary surgery.
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Insulin autoimmune syndrome is characterized by spontaneous hypoglycemia, high concentrations of serum immunoreactive insulin, and the presence of autoantibodies to insulin without previous insulin injection. A 71-year-old woman with diabetes, who had been treated with oral hypoglycemic agents, suffered from frequent hypoglycemia. High insulin levels and the presence of insulin autoantibodies were shown, so insulin autoimmune syndrome was diagnosed. Drugs containing sulfhydryl groups play an important role in the pathogenesis of insulin autoimmune syndrome. Alpha-lipoic acid, which contains a sulfhydryl group, was administered before the onset of hypoglycemia. The patient's human leukocyte antigen (HLA) type was HLA-Cw4 and DRB1*0406. There is a strong correlation between HLA-DRB1*0406 and insulin autoimmune syndrome. The patient was treated with prednisolone and has not had a hypoglycemic attack since. We report a case of insulin autoimmune syndrome, possibly associated with alpha-lipoic acid.
Subject(s)
Aged , Female , Humans , Autoantibodies , HLA-C Antigens , HLA-DRB1 Chains , Hypoglycemia , Hypoglycemic Agents , Insulin , Leukocytes , Prednisolone , Thioctic AcidABSTRACT
Insulin autoimmune syndrome is an uncommon cause of hypoglycemia. According to the type of antibody, it can be classified as caused by insulin or insulin receptor autoantibodies. Generally, insulin autoimmune syndrome develops following exposure to exogenous insulin or sulfhydryl medications, although insulin or insulin receptor antibody may also occur spontaneously. We treated a 54-year-old woman who developed spontaneous hypoglycemia. The patient had repeated hypoglycemia despite the infusion of dextrose solution. Her serum insulin, c-peptide, and insulin autoantibody were elevated, even during the hypoglycemic periods. Insulin receptor autoantibody and HLA-cw4/B62/DR4 were positive. After steroid and diazoxide treatment, the hypoglycemic symptoms improved gradually. No further hypoglycemic episodes occurred after tapering the medication over 1 year. We present a case of insulin autoimmune syndrome with positive insulin and insulin receptor autoantibodies.
Subject(s)
Female , Humans , Middle Aged , Autoantibodies , C-Peptide , Diazoxide , Glucose , Hypoglycemia , Insulin , Receptor, InsulinABSTRACT
OBJECTIVE: To provide reference for the treatment of drug-induced insulin autoimmune syndrome (IAS). METHODS: One drug-induced IAS case reported in our hospital was analyzed, combining with clinical information of reported drug-induced IAS cases in China. RESULTS: Of reported drug-induced IAS cases in China, those etiology and symptoms were correspond with the characteristics of foreign reported IAS cases; patients’ age was lower than the average age of IAS patients; IAS patients were mostly treated with glucocorticoid in China, which is significantly different from reported cases in Japan. CONCLUSION: Most IAS patients are in serious condition. In clinical practice pharmacists should fully understand the medication history of patients with spontaneous hypoglycemia, and identify the difference between IAS and spontaneous hypoglycemia carefully to reduce the pain and risk of death for patients resulted from misdiagnosis and missed diagnosis.