Insulin Autoimmune Syndrome (Hirata Disease): Case Report in a Caucasian Patient with New-Onset Diabetes

Sudano, Maurizio; Turchi, Federica; Sossai, Paolo

doi:10.5923/j.cmd.20120205.02

Cited by 5 publications

(4 citation statements)

References 11 publications

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“…The low prevalence of IAS among Caucasians can be explained by the low prevalence of DRB1*0406 in this population (9). One Bulgarian person with IAS reported to have DRB1*04:03, DRB1*15 (10) and a 7-year-old Brazilian women with IAS syndrome was reported to have HLA -DRB1*1104 genotype (11). There is no report showing HLA B1*03 and B3*02 and DRB1*14 (as detected in our case) in Hirata syndrome.…”

Section: Discussionmentioning

confidence: 99%

Insulin Autoimmune Syndrome: a rare cause of postprandial hypoglycemia

Sahni

Trivedi

Omer

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryA 65-year-old obese Caucasian woman presented with symptomatic postprandial hypoglycemic episodes, resolution of symptoms with carbohydrate intake and significantly elevated anti-insulin antibody levels. She did not have any evidence for the use of oral antidiabetic medications, insulin, herbal substances, performing strenuous exercise or history of bariatric surgery. Fingerstick blood glucose readings revealed blood sugar of 35 mg/dL and 48 mg/dL, when she had these symptoms. Her medical history was significant for morbid obesity, hypothyroidism and gastro esophageal reflux disease. Her home medications included levothyroxine, propranolol and omeprazole. A blood sample obtained during the symptoms revealed the following: fingerstick blood sugar 38 mg/dL, venous blood glucose 60 mg/dL (normal (n): 70–99 mg/dL), serum insulin 202 IU/mL (n: <21), proinsulin 31.3 pmol/L (n: <28.9), C-peptide 8 ng/mL (n: 0.9–7), beta-hydroxybutyrate 0.12 mmol/L (n: 0.02–0.27) anti-insulin antibody >45.4 U/mL (n: <0.4). The result obtained while screening for serum sulfonylurea and meglitinides was negative. The repeated episodes of postprandial hypoglycemia associated with significantly elevated anti-insulin antibodies led to a diagnosis of insulin antibody syndrome (IAS). Significant improvement of hypoglycemic symptoms and lower anti-insulin antibody levels (33 U/mL) was noted on nutritional management during the following 6 months. Based on a report of pantoprazole-related IAS cases, her omeprazole was switched to a H2 receptor blocker. She reported only two episodes of hypoglycemia, and anti-insulin antibody levels were significantly lower at 10 U/mL after the following 12-month follow-up.Learning points:Initial assessment of the Whipple criteria is critical to establish the clinical diagnosis of hypoglycemia accurately.Blood sugar monitoring with fingerstick blood glucose method can provide important information during hypoglycemia workup.Autoimmune hypoglycemia is a rare cause of hypoglycemia, which can be diagnosed on high index of clinical suspicion and systematic evaluation.

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Section: Discussionmentioning

confidence: 99%

Insulin Autoimmune Syndrome: a rare cause of postprandial hypoglycemia

Sahni

Trivedi

Omer

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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“…insulinoma, critical illnesses, and endocrine deficiencies). [12] IAS is characterized by the presence of an autoantibody to native insulin. The autoantibody stays in the blood and binds to insulin.…”

Section: Discussionmentioning

confidence: 99%

Insulin autoimmune syndrome (Hirata Disease): Case report in a saudi female patient with graves' disease and literature review

Almutairi

Aljumah

2020

Journal of Diabetes and Endocrine Practice

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Uncommon scenario of spontaneous hypoglycemia after starting carbimazole due to insulin autoimmune syndrome, characterized by high levels of insulinemia and circulating autoantibodies to insulin without prior insulin administration. So far, in Western countries, <70 cases have been published. In addition, more than 380 cases of this syndrome have been reported.

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“…In conclusion, new informative cases of IAS in Caucasians continued to appear in the world literature [27,28,69,70,75,[90][91][92][93][94][95][96][97][98][99]. The very wide range of their clinical presentations and biochemical findings re-emphasised that IAS is a highly heterogeneous syndrome.…”

Section: Comments and Conclusionmentioning

confidence: 98%

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

Ismail¹

2016

Clinical Chemistry and Laboratory Medicine (CCLM)

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Abstract:Insulin 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.Keywords: endogenous insulin antibodies; Hirata's disease, insulin autoimmune hypoglycaemia; hypoglycaemia in non-diabetic; insulin autoimmune syndrome (IAS); interference in immunoassays of insulin, proinsulin and C-peptide; wrong immunoassay results .

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Insulin Autoimmune Syndrome (Hirata Disease): Case Report in a Caucasian Patient with New-Onset Diabetes

Cited by 5 publications

References 11 publications

Insulin Autoimmune Syndrome: a rare cause of postprandial hypoglycemia

Insulin Autoimmune Syndrome: a rare cause of postprandial hypoglycemia

Insulin autoimmune syndrome (Hirata Disease): Case report in a saudi female patient with graves' disease and literature review

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

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