scholarly journals Primary insulin autoimmune syndrome in an Italian woman: a case report

2015 ◽  
Vol 9 (2) ◽  
pp. 169 ◽  
Author(s):  
Antonio Balestrieri ◽  
Elena Magnani ◽  
Cecilia Ragazzini ◽  
Giampiero Pasini
Keyword(s):  
Case Report ◽  
Insulin Antibodies ◽  
Total Serum ◽  
Immunoreactive Insulin ◽  
High Concentration ◽  
Hematological Diseases ◽  
Italian Woman ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Primary Form

Insulin autoimmune syndrome (IAS) is a rare syndrome characterized by fasting or postprandial hypoglycemia, high levels of anti-insulin antibodies and high concentration of total serum immunoreactive insulin. It is relatively known in Japan, rare in remaining Asia and it is extremely uncommon in Western countries, being characterized by a different race-related incidence and associated with HLADR4 alleles. Usually IAS is related to particular drugs, or to autoimmune, rheumatologic or hematological diseases, while it is very rare as a primary form. Here we described a case of an Italian woman affected by a primary form of Hirata syndrome.

scholarly journals Insulin autoimmune syndrome: case report

2004 ◽  
Vol 122 (4) ◽  
pp. 178-180 ◽  
Author(s):  
Rodrigo Oliveira Moreira ◽  
Giovanna Aparecida Balarini Lima ◽  
Patrícia Carla Batista Peixoto ◽  
Maria Lucia Fleiuss Farias ◽  
Mario Vaisman
Keyword(s):  
Case Report ◽  
Pancreatic Tumor ◽  
Histopathological Examination ◽  
Severe Trauma ◽  
Insulin Antibodies ◽  
Insulin Levels ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
High Insulin ◽  
Biochemical Evaluation

CONTEXT: Insulin autoimmune syndrome (IAS, Hirata disease) is a rare cause of hypoglycemia in Western countries. It is characterized by hypoglycemic episodes, elevated insulin levels, and positive insulin antibodies. Our objective is to report a case of IAS identified in South America. CASE REPORT: A 56-year-old Caucasian male patient started presenting neuroglycopenic symptoms during hospitalization due to severe trauma. Biochemical evaluation confirmed hypoglycemia and abnormally high levels of insulin. Conventional imaging examinations were negative for pancreatic tumor. Insulin antibodies were above the normal range. Clinical remission of the episodes was not achieved with verapamil and steroids. Thus, a subtotal pancreatectomy was performed due to the lack of response to conservative treatment and because immunosuppressants were contraindicated due to bacteremia. Histopathological examination revealed diffuse hypertrophy of beta cells. The patient continues to have high insulin levels but is almost free of hypoglycemic episodes.

scholarly journals Insulin autoimmune syndrome in an occidental woman: a case report and literature review

2018 ◽  
Vol 62 (5) ◽  
pp. 566-570 ◽  
Author(s):  
Mariella Zaiden Rezende Reis ◽  
Virgínia Oliveira Fernandes ◽  
Eveline Gadelha Pereira Fontenele ◽  
Ana Paula Abreu Martins Sales ◽  
Renan Magalhães Montenegro ◽  
...  
Keyword(s):  
Case Report ◽  
Literature Review ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome

Reactive hypoglycemic coma due to insulin autoimmune syndrome: Case report and literature review

1992 ◽  
Vol 92 (6) ◽  
pp. 681-685 ◽  
Author(s):  
Henry B. Burch ◽  
Stephen Clement ◽  
Michael S. Sokol ◽  
Frank Landry
Keyword(s):  
Case Report ◽  
Literature Review ◽  
Hypoglycemic Coma ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome

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.

scholarly journals Insulin autoimmune syndrome as a cause of recurrent hypoglycemia in a carbimazole user: a case report from Nepal

2019 ◽  
Vol Volume 12 ◽  
pp. 29-32 ◽  
Author(s):  
Vivek Pant ◽  
Bijay Bhandari ◽  
Suman Baral ◽  
Sangha Ratna Bajracharya
Keyword(s):  
Case Report ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Recurrent Hypoglycemia

Gliclazide-Induced Insulin Autoimmune Syndrome: A Rare Case Report and Review on Literature

2017 ◽  
Vol 16 (4) ◽  
pp. 230-234 ◽  
Author(s):  
Xiu Feng ◽  
Lu Yuan ◽  
Yun Hu ◽  
Yunqing Zhu ◽  
Fan Yang ◽  
...  
Keyword(s):  
Case Report ◽  
Rare Case ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Rare Case Report

Insulin Autoimmune Syndrome After Exposure to Clopidogrel: A Case Report

2020 ◽  
Vol 20 (8) ◽  
pp. 1355-1362 ◽  
Author(s):  
Yihong Jiang ◽  
Lihua Wang ◽  
Fanghong Shi ◽  
Huan Zhou ◽  
Jun Zheng ◽  
...  
Keyword(s):  
Case Report ◽  
Multidisciplinary Team ◽  
Evaluation Process ◽  
First Episode ◽  
C Peptide ◽  
Prednisone Therapy ◽  
Insulin Autoimmune Syndrome ◽  
Autoimmune Syndrome ◽  
Insulin Autoantibody ◽  
Sh Group

Background: Insulin autoimmune syndrome (IAS) is a rare cause of hypoglycemia that is characterized by hyperinsulinemia, hypoglycemia, and a high autoantibody titer. About 50% of patients with IAS have taken a medication containing sulfhydryl (-SH) groups. We present a case of IAS that developed after taking clopidogrel, a drug with an active metabolite that contains an SH-group. Case report: IAS was suspected in a 63-year-old Chinese man because of high concentrations of insulin and C-peptide during hypoglycemic episodes, and positivity for anti-insulin autoantibody (IAA). During his first episode of hypoglycemia, no trigger medication was identified and prednisone therapy was effective. However, imaging examination revealed a colonic carcinoma and the patient was discharged to undergo surgery. He had no episodes of hypoglycemia for 10 weeks after discontinuation of the prednisone, but then hypoglycemia recurred. A review of his medication revealed that he had taken a 10-day course of clopidogrel just before the recurrence. Therefore, a specialized multidisciplinary team consisting of endocrinologists, dieticians, and clinical pharmacists took charge of his management. Prednisone therapy was restarted and tapered off over 16 weeks. The patient also consumed small, frequent, low-carbohydrate meals and was instructed to avoid trigger medications. No further episodes of hypoglycemia were detected. His insulin and C-peptide concentrations and his anti-IAA index normalized during the follow-up period. Conclusion: SH-group-containing drugs might induce or exacerbate hypoglycemia in patients with a history of IAS. Furthermore, patients with IAS can benefit from multidisciplinary team management. We suggest herein an evaluation process for patients suspected of IAS.

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.

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