scholarly journals Severe Fasting Hypoglycemia Mimicking Insulinoma in Three Patients With Insulin Autoimmune Syndrome

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A399-A399
Author(s):  
Alessandro Rossini ◽  
Sara Cassibba ◽  
Cristiana Scaranna ◽  
Anna Corsi ◽  
Rosalia Bellante ◽  
...  
Keyword(s):  
Clinical Manifestations ◽  
Glycemic Variability ◽  
Insulin Binding ◽  
Glucose Levels ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Therapeutic Procedures ◽  
History Of ◽  
Fasting Hypoglycemia ◽  
Sulphydryl Compound

Abstract Insulin autoimmune syndrome (IAS) is a rare cause of hypoglycemia characterized by the presence of insulin autoantibodies (IAA) in patients without prior exposure to exogenous insulin. Differential diagnosis with other causes of hypoglycemia may be complex. We report three IAS cases with severe fasting hypoglycemia, referred to our Unit for the diagnostic workup of insulinoma. All three patients (two women and a man, age 66, 44, and 50 years) had history of severe fasting hypoglycemia leading to loss of consciousness along with weight gain. Both insulin and C-peptide were high, but their levels varied greatly among patients, ranging from 24 to 1500 μU/ml (n.v. <16.3) and from 11 to 27 ng/mL (n.v. < 4,2), respectively. Imaging studies for insulinoma were negative. In all patients, evidence of elevated IAA (ranging from 310 to 660 UA, n.v. < 5) allowed diagnosis of IAS. Two patients were taking alpha lipoic acid, a sulphydryl compound consistently associated to IAS, while in the other the HLA-DRB1*0403 haplotype, conferring susceptibility to IAS, was detected. Continuous monitoring glucose (CGM) (iPro2; Medtronic Diabetes, CA, USA) showed in all patients the presence of prolonged hypoglycemia (with time spent with blood glucose below 54 mg/dL ranging from 9 to 20% of total monitoring time), and in one case the coexistence of high glucose levels after meals. One patient responded well to diazoxide treatment, while the others required both chronic steroid therapy and the use of plasmaphereses. Conclusion: Clinical manifestations of IAS vary widely among patients, without a direct correlation between symptoms severity and levels of both insulin and IAA; prandial hyperglycemia may also be present, leading to increases in glycated hemoglobin. Our patients displayed severe fasting hypoglycemic attacks that initially posed the suspicion of insulinoma. The assessment of IAA is thus mandatory in cases of fasting hypoglycemia, before proceeding to more expensive and probably unnecessary diagnostic and therapeutic procedures. CGM is a useful tool in evaluation and management of IAS, allowing the assessment of hypoglycemia duration and the detection of the wide glycemic variability secondary to the complex mechanism of insulin binding to IAA.

scholarly journals Glycemic Variability in Diabetes Increases the Severity of Influenza

mBio ◽  
2020 ◽  
Vol 11 (2) ◽  
Author(s):  
Rebecca J. Marshall ◽  
Pornthida Armart ◽  
Katina D. Hulme ◽  
Keng Yih Chew ◽  
Alexandra C. Brown ◽  
...  
Keyword(s):  
Blood Glucose ◽  
Glycemic Variability ◽  
Endothelial Barrier ◽  
Glucose Levels ◽  
Blood Glucose Levels ◽  
Severe Influenza ◽  
Increased Risk ◽  
History Of

ABSTRACT People with diabetes are two times more likely to die from influenza than people with no underlying medical condition. The mechanisms underlying this susceptibility are poorly understood. In healthy individuals, small and short-lived postprandial peaks in blood glucose levels occur. In diabetes mellitus, these fluctuations become greater and more frequent. This glycemic variability is associated with oxidative stress and hyperinflammation. However, the contribution of glycemic variability to the pathogenesis of influenza A virus (IAV) has not been explored. Here, we used an in vitro model of the pulmonary epithelial-endothelial barrier and novel murine models to investigate the role of glycemic variability in influenza severity. In vitro, a history of glycemic variability significantly increased influenza-driven cell death and destruction of the epithelial-endothelial barrier. In vivo, influenza virus-infected mice with a history of glycemic variability lost significantly more body weight than mice with constant blood glucose levels. This increased disease severity was associated with markers of oxidative stress and hyperinflammation both in vitro and in vivo. Together, these results provide the first indication that glycemic variability may help drive the increased risk of severe influenza in people with diabetes mellitus. IMPORTANCE Every winter, people with diabetes are at increased risk of severe influenza. At present, the mechanisms that cause this increased susceptibility are unclear. Here, we show that the fluctuations in blood glucose levels common in people with diabetes are associated with severe influenza. These data suggest that glycemic stability could become a greater clinical priority for patients with diabetes during outbreaks of influenza.

scholarly journals Hypoglycemia due to insulin autoimmune syndrome (Hirata’s disease): a rare cause of hypoglycaemia

2021 ◽  
Vol 8 (8) ◽  
pp. 1220
Author(s):  
Het V. Patel ◽  
Sunil Kumar ◽  
Kalpesh Moradiya ◽  
Vidhi Shah
Keyword(s):  
Nutrition Therapy ◽  
Serum Glucose ◽  
Insulin Antibodies ◽  
Laboratory Evaluation ◽  
Hyperinsulinemic Hypoglycemia ◽  
Lifestyle Modifications ◽  
Glucose Levels ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Recurrent Hypoglycaemia

Although the most common cause of recurrent hypoglycaemia is diabetes mellitus as patient is on antidiabetic medications which can be prevented by modification of antidiabetic doses, nutrition therapy and lifestyle modifications. Some endogenous hyperinsulinemic conditions like insulinoma, functional beta cell disorders and insulin autoimmune syndromes, hormonal deficiencies can cause serious and sometimes life threatening hypoglycaemia. So further laboratory evaluation like plasma/serum glucose levels, c-peptide levels, insulin levels, insulin antibodies and imaging studies are needed to evaluate unexplained hypoglycaemia. Here we report a case of insulin autoimmune syndrome in a 67 year old Indian male who had presented to us with multiple episodes of spontaneous hypoglycaemia. On further workup, the patient was found to have endogenous hyperinsulinemic hypoglycemia. As the patient’s abdominal imaging revealed no apparent cause of EHH, on further evaluation he came positive for insulin antibodies. Patient was diagnosed as IAS and he was given frequent small meals and complex carbohydrate diet and he had improved symptomatically. The incidence of IAS is most common in Japan and very few cases have been reported from India, so it should be kept in differential diagnosis of recurrent hypoglycaemia.

The insulin autoimmune syndrome (IAS) as a cause of hypoglycaemia: an update on the pathophysiology, biochemical investigations and diagnosis

2016 ◽  
Vol 54 (11) ◽  
Author(s):  
Adel A.A. Ismail
Keyword(s):  
Glycaemic Control ◽  
Clinical Manifestations ◽  
Insulin Antibodies ◽  
Pancreatic Hormones ◽  
Insulin Mimetic ◽  
Insulin Autoimmune Syndrome ◽  
Endogenous Insulin ◽  
Autoimmune Syndrome ◽  
Potential Impact ◽  
Receptor Antibodies

AbstractInsulin autoimmune syndrome (IAS) is considered to be very rare in Caucasians. Understanding its pathophysiology is paramount in (a) appreciating its potential impact on analyses of pancreatic hormones and (b) explaining its highly variable clinical manifestations in non-diabetic, non-acutely ill patients with indeterminate hypoglycaemia. The underlying aetiology of IAS is the presence of variable affinity/avidity endogenous insulin antibodies in significant amounts. The two types of insulin antibodies namely antibodies which bind insulin and/or proinsulin(s) and receptor antibodies (insulin mimetic) will be discussed. Their biochemical and immunological roles in causing hypoglycaemia will be highlighted. Clinical manifestations of IAS can vary from mild and transient to spontaneous, severe and protracted hypoglycaemia necessitating in extreme cases plasmapheresis for glycaemic control. Antibodies of IAS can interfere in pancreatic immunoassay tests causing erroneous and potentially misleading results. Thorough testing for endogenous insulin antibodies must be considered in the investigations of non-diabetic, non-acutely ill patients with indeterminate and/or unexplained hypoglycaemia.

scholarly journals “The Unusual Suspect” - Insulin Autoimmune Syndrome, an Extremely Rare Cause of Hypoglycemia

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A355-A355
Author(s):  
Hassaan B Aftab ◽  
Carl D Malchoff
Keyword(s):  
Insulin Antibody ◽  
Diabetic Patients ◽  
High Dose ◽  
Blood Draw ◽  
Family History Of Diabetes ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Asian Descent ◽  
History Of ◽  
Antibody Titers

Abstract Background: Insulin autoimmune syndrome (IAS), also called Hirata syndrome, is a rare diagnosis which is characterized by hypoglycemia, normal or elevated fasting insulin level, and elevated anti-insulin antibody titers. Clinical Case: 45-year-old nondiabetic Caucasian female presented to the emergency department (ED) with chief complaint of near-syncope. She reported difficulty concentrating and feeling faint roughly 1 hour after consuming breakfast. Upon ED arrival, fingerstick glucose (FSG) was 47 mg/dL. Her symptoms resolved with ingestion of orange juice and repeat blood glucose (BG) 15 minutes later was 120 mg/dL. She reported similar symptoms in the last 5 years happening “every few months” without a predictable pattern; twice with witnessed loss of consciousness. Workup for syncope in the past included normal neurologic, cardiac, and tilt-table testing. She had no prior low FSG/BG recorded, no personal history of bariatric surgery or family history of diabetes mellitus and no access to antihyperglycemic medications. Past medical history included ulcerative colitis and medications included sulfasalazine and multivitamins. Pertinent review of systems was negative for weight loss, nausea, and diarrhea. Vital signs were within normal limits. Physical exam was unremarkable. Height was 165 cm, weight 67.8 kg and body mass index 24.9 kg/m2. Blood draw after overnight fast revealed BG of 78 mg/dL, C-peptide 0.7 ng/mL (ref range 0.8–3.5), insulin 10 µIU/mL (3–19), proinsulin <1.6 pmol/L (<8.0), insulin-like growth factor-2 (IGF-2) 339 ng/mL (180–580) and insulin antibody level 10.0 U/mL (0.0–0.4). She was managed conservatively with frequent small meals and patient has continued to do well without recurrence of symptoms. Conclusion: IAS is an exceedingly rare disease entity with about 460 cases described in literature, mostly in patients of Asian descent. Workup for hypoglycemia in non-diabetic patients should include insulin antibody titers to rule out IAS. There appears to be a genetic predisposition to developing IAS in patients with major histocompatibility complex (MHC) genes HLA-DQA1, HLA-DQB1 and HLA-DRB1. It is now being more frequently described in patients of non-Asian descent, particularly those with other autoimmune or plasma cell dyscrasias. It is also associated with drugs containing sulfhydryl groups and viral infections. Treatment should be tailored to severity of disease. Most drug induced IAS resolves after cessation of culprit drug. Conservative management is usually the first step with frequent, low carbohydrate meals with diazoxide or octreotide as adjunctive options. Immunosuppression (high-dose prednisolone or rituximab) and plasma exchanged are reserved severe or refractory cases.

scholarly journals MON-686 Insulin Autoimmune Syndrome as a Rare Cause of Hypoglycemia

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Marwa Mokhtar ◽  
Wajiha Gul ◽  
Monica Skarulis
Keyword(s):  
Pancreatic Head ◽  
Acute Onset ◽  
Insulin Antibodies ◽  
Hyperinsulinemic Hypoglycemia ◽  
Short Term Treatment ◽  
Male Physician ◽  
C Peptide ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
History Of

Abstract Background: Insulin Autoimmune Syndrome (IAS) or Hirata disease is reported more in Asians. We report the case of a patient with acute onset hypoglycemia secondary to IAS. Case Presentation: A 64 year old ARAB male physician presented with 2 weeks history of episodic hypoglycaemia with blood glucose (BG) 40–60 mg/dl, unrelated to food and occurring throughout the day. His attacks were associated with sweating, dizziness and palpitations, relieved by intake of juice. He reported 3 kg weight gain since the beginning of his symptoms. Past medical history revealed hypertension, past HCV infection with negative PCR and history of allergy to sulfa drugs. His medications included bisoprolol 2.5 mg daily. Physical exam showed no acanthosis nigericans. Laboratory investigations: Normal hepatic, renal and thyroid functions. 72 Hours fasting test was performed. at hour 3, the BG was 2.27 mmol (40.86 mg/dl), insulin > 1000 mU/ml (N: 2.6–24.9), c- peptide 15 ng/ml (N: 1.1–4.4), negative beta-hydroxybutyrate, with good glycemia response to glucagon 1mg. Urine sulfonylurea (SU) screen was negative. Insulin antibodies titre was high 8.9 nmol/L (N: 0–0.02) and Proinsulin >700 pmol/L (N: 3.6–22). MRI Abdomen showed only 3 mm lesion in the pancreatic head, likely a cyst. During hospitalization, the patient continued to have episodic hypoglycaemia and received dextrose 10% and octreotide injection. Bisoprolol was changed to amlodipine as it may mask his symptoms. Episodes of hypoglycaemia improved with octreotide but did not resolve completely. Prednisolone 30 mg daily was started with improvement and was tapered slowly after 16 weeks after mild hyperglycemia developed. Repeat work up showed reduction in insulin 67.4 mcunit/ml, C- peptide 2.66 ng/ml and insulin antibody titres to 0.24 nmol/L and remission of hypoglycemia. Discussion: Autoimmune hypoglycaemia is rare resulting from insulin antibodies(IAS), or anti-insulin receptor antibodies (Type B insulin Resistance). IAS can be triggered by drugs or viruses including hepatitis C and is associated with autoimmune diseases and hematologic malignancies. the differential diagnosis of hyperinsulinemic hypoglycemia include insulinoma, the presence of extreme levels of insulin, c-peptide and proinsulin and insulin antibodies is diagnostic of AIS. Short-term treatment with steroids was effective in treating hypoglycaemia and careful follow-up is ongoing. If the patient recurs, rituximab will be employed. References: 1-CHURCH, D., CARDOSO, L., KAY, R. G., WILLIAMS, C. L., FREUDENTHAL, B., CLARKE, C., HARRIS, J., MOORTHY, M., KARRA, E., GRIBBLE, F. M., REIMANN, F., BURLING, K., WILLIAMS, A. J. K., MUNIR, A., JONES, T. H., FUHRER, D., MOELLER, L. C., COHEN, M., KHOO, B., HALSALL, D. & SEMPLE, R. K. 2018. Assessment and Management of Anti-Insulin Autoantibodies in Varying Presentations of Insulin Autoimmune Syndrome. J Clin Endocrinol Metab, 103, 3845–3855.

Insulin autoimmune syndrome (Hirata’s disease) in an Italian patient: a case report and review of the literature

2018 ◽  
Vol 56 (6) ◽  
pp. 889-895 ◽  
Author(s):  
Simona Censi ◽  
Maria Paola Albergoni ◽  
Nicoletta Gallo ◽  
Mario Plebani ◽  
Marco Boscaro ◽  
...  
Keyword(s):  
Blood Glucose ◽  
Glucose Tolerance ◽  
Good Control ◽  
Oral Glucose ◽  
High Blood Glucose ◽  
Glucose Levels ◽  
Insulin Autoimmune Syndrome ◽  
Blood Glucose Levels ◽  
Autoimmune Syndrome ◽  
Insulin Autoantibody

Abstract We describe the case of a 54-year-old Caucasian Italian male experiencing episodes of hypoglycemia, occurring mainly after meals. He had never been exposed to insulin and was taking ramipril, flecainide and acetylsalicylic acid. An oral glucose tolerance test (OGTT) showed high blood glucose levels diagnostic for diabetes mellitus at 120 min and hypoglycemia with inappropriately high insulin levels at 240 min. The 72-h fasting test, abdominal computed tomography (CT) and positron emission tomography-CT were normal. Insulin autoantibodies were positive at high titers, prompting a diagnosis of insulin autoimmune syndrome (IAS). The patient was advised to take frequent, small meals and thus achieved a good control of his hypoglycemic symptoms. After 18 months of this dietary management, his insulin autoantibody levels decreased considerably but remained detectable. During an OGTT, his blood glucose levels at 120 min were now indicative of an impaired glucose tolerance rather than diabetes, and there was improvement in the glucose nadir. The patient had no other clinical or latent autoimmune diseases. Here we discuss the main features of IAS (also known as Hirata’s disease) and review the cases of IAS reported in Italy to date.

scholarly journals Insulin Autoimmune Syndrome in a 25-Year-Old, Previously Healthy Kuwaiti Man

2019 ◽  
Vol 2019 ◽  
pp. 1-4 ◽  
Author(s):  
Einas Alrashidi ◽  
Thamer Alessa
Keyword(s):  
Anabolic Steroids ◽  
Insulin Level ◽  
Complete Recovery ◽  
Insulin Analogues ◽  
Hyperinsulinaemic Hypoglycaemia ◽  
Insulin Autoimmune Syndrome ◽  
Endogenous Insulin ◽  
Autoimmune Syndrome ◽  
History Of ◽  
Work Up

Insulin autoimmune syndrome (IAS) is a disease characterized by hyperinsulinaemic hypoglycaemia associated with autoantibodies against endogenous insulin. We have described a case of a 25-year-old, previously healthy Kuwaiti man who was admitted to the Mubarak Al-Kabeer hospital with a history of recurrent hypoglycaemia. The patient revealed that he had taken several different injectable anabolic steroids and growth hormone with oral amino acids and other tablets (fat burners) for bodybuilding in the last two months. He denied knowingly using insulin or insulin analogues. The patient had elevated fasting insulin level (>301 uIU/mL) and elevated insulin autoantibodies (>100.0 IU/mL). After appropriate work-up, he was diagnosed with IAS. After treatment with prednisolone (1 mg/kg/day), the patient had complete recovery. In patients with repeated hypoglycaemia, IAS should be considered in the differential diagnosis. Glucocorticoid therapy can be effective for the treatment of hypoglycaemia in patients with IAS.

Arare case of caucasian patient with insulin autoimmune syndrome induced by a-lipoic acid

2020 ◽  
Author(s):  
Spyridoula Glikofridi ◽  
Labrini Papanastasiou ◽  
Athina Markou ◽  
Vasileios Andrianesis ◽  
Christos Gravvanis ◽  
...  
Keyword(s):  
Lipoic Acid ◽  
Caucasian Patient ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome

Study of Diabetic Mellitus and Knowledge of Lotion Foot Care on the Community

2018 ◽  
Vol 9 (3) ◽  
Author(s):  
Hariyadi DM ◽  
Athiyah U ◽  
Hendradi E ◽  
Rosita N ◽  
Erawati T ◽  
...  
Keyword(s):  
Blood Glucose ◽  
T Test ◽  
Small Scale ◽  
Scale Production ◽  
Foot Care ◽  
Diabetic Mellitus ◽  
Glucose Levels ◽  
Blood Glucose Levels ◽  
Before And After ◽  
History Of

The prevention of Diabetic Mellitus (DM) and its complications is the main aim of this study, in addition to the training of lotion foot care application and the development of small scale industry. The research team delivered knowledge in the form of training on Diabetic Mellitus, healthy food, treatment and prevention of complications, and small-scale production of cosmetic products. The aim of this study was to determine the correlation between training on diabetic and lotion foot care application as preventive measures against diabetic complications on the patient's blood glucose levels in the community of residents in Banyuurip Jaya, Surabaya. It was expected from this training that the knowledge of the residents increases and people living with diabetic undergo lifestyle changes and therefore blood sugar levels can be controlled. The parameters measured in this research were blood glucose levels, the anti diabetic drug types consumed, and compliance on diabetics. This study used the data taken from 60 patients with DM over a period of one month. Questionnaires and log books was used to retrieve data and changes in blood glucose levels in diabetic patients. The results showed the demographic data of patients with type 2 diabetic of 85% female and 15% male, with the range of patients aged of 61-70 years of 46.67% and had history of diabetic (90%). The history of drugs consumed by respondents was anti diabetic drugs such as metformin (40%), glimepiride (33.37%) and insulin (6.67%). In addition, the increased knowledge of DM patients after being given the training compared to before training was shown in several questions in the questionnaire. A statistical analysis using t-test analyzed a correlation between training provided in order to enhance understanding of the patient, as well as correlation with blood glucose levels. A paired T-test showed that there was a relationship between the knowledge of trainees before and after training (p less than 0.05). An interesting result was that there was no relationship between blood glucose levels before and after training provided (p> 0.05).

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