Insulin Autoimmune Syndrome after the Third Therapy with Methimazole.

Hakamata, Mutsumi; Itoh, Mitsuyasu; Sudo, Y; Miyata, Norikazu

doi:10.2169/internalmedicine.34.410

Cited by 5 publications

(3 citation statements)

References 11 publications

(7 reference statements)

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“…22 Yet, there are reports of drug-induced IAS developing years after the first administration of the offending medication, due to an additional and subsequent cycle of the medical therapy. 39 Many viral infections have been reported to be triggers for the development of IAS: measles virus, mumps virus, rubella virus, varicella zoster virus, coxsackie B virus and hepatitis C virus. 77 The supposed mechanism is that the viral infection acts as a super-antigen, thus triggering the production of the IAA that cause the syndrome.…”

Section: Etiologymentioning

confidence: 99%

<p>Insulin Autoimmune Syndrome (Hirata Disease): A Comprehensive Review Fifty Years After Its First Description</p>

Cappellani¹,

Macchia²,

Falorni³

et al. 2020

DMSO

114

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Insulin autoimmune syndrome (IAS), also named Hirata's disease, is a rare condition characterized by hypoglycemic episodes due to the presence of high titers of insulin autoantibodies (IAA). IAS is a form of immune-mediated hypoglycemia, which develops when a triggering factor (ie, a medication or a viral infection) acts on an underlying predisposing genetic background. IAS pathogenesis involves the formation of insulin-IAA complexes that induce glycemic alterations with a double-phase mechanism: IAA prevent insulin to bind its receptor in the postprandial phase, possibly resulting in mild hyperglycemia; thereafter, insulin is released from the complexes irrespective of blood glucose concentrations, thus inducing hypoglycemia. The diagnosis of IAS is challenging, requiring a careful workup aimed at excluding other causes of hyperinsulinemic hypoglycemia. The gold standard for the definitive diagnosis is the finding of IAA in a blood sample. Because IAS is frequently a self-remitting disease, its management mostly consists of supportive measures, such as dietary modifications, aimed at preventing the development of hypoglycemia. Pharmacological therapies may occasionally be necessary for patients presenting with severe manifestations of IAS. Available therapies may include drugs that reduce pancreatic insulin secretion (somatostatin analogues and diazoxide, for instance) and immunosuppressive agents (glucocorticoids, azathioprine and rituximab). The purpose of this review is to provide a comprehensive analysis of the disease, by describing the burden of knowledge that has been obtained in the 50 years following its first description, took in 1970, and by highlighting the points that are still unclear in its pathogenesis and management.

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Section: Etiologymentioning

confidence: 99%

<p>Insulin Autoimmune Syndrome (Hirata Disease): A Comprehensive Review Fifty Years After Its First Description</p>

Cappellani¹,

Macchia²,

Falorni³

et al. 2020

DMSO

114

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“…A case report describes a patient Graves’ disease who had been treated with methimazole 2 years prior to the onset of hypoglycemic attacks. In this case the attacks had begun after the third course of methimazole therapy [ 8 ].…”

Section: Discussionmentioning

confidence: 99%

Uncommon triggers of insulin autoimmune syndrome: a case report

Abeyagunawardena,

Madurapperuma,

Thuvarakan

et al. 2024

J Med Case Reports

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Background Insulin autoantibody syndrome (IAS), or Hirata disease, is caused by high concentrations of insulin autoantibodies, which result in spontaneous, mainly post-prandial, hypoglycemic episodes. We report a case of a previously healthy 67-year-old man presenting with recurrent fasting hypoglycemia culminating in a diagnosis of insulin autoimmune syndrome linked to omeprazole and probably spices, namely, coriander, and ginger. Case presentation A previously healthy 67-year-old Sinhalese man presented with recurrent syncopal attacks for 3 months, which were found to be hypoglycemic episodes. He experienced mainly fasting hypoglycemic attacks, at a frequency gradually increasing to daily attacks. His cardiovascular, respiratory, abdominal, and neurologic examinations were normal. He was found to have insulin levels > 6000 mU/L and a post-polyethylene glycol insulin recovery of less than 9.5%. Contrast-enhanced computed tomography of the pancreas was normal. The diagnosis of insulin autoantibody syndrome was confirmed by testing for the insulin autoantibody level, yielding a level of > 300 U/mL. With regard to a possible trigger, he had a history of omeprazole intake for 2 weeks, 4 weeks prior to the onset of symptoms. He also consumed an herbal supplement containing coriander and ginger extracts daily for a period of 1 year, approximately 2 years prior to the onset of hypoglycemic attacks. He was commenced on prednisolone 30 mg daily, and hypoglycemic episodes responded dramatically, and thus he was tapered off corticosteroids. Conclusion Omeprazole-induced insulin autoantibody syndrome is likely in this patient; however, the known hypoglycemic effects of coriander and ginger make it worthwhile to consider a possible association with insulin autoantibody syndrome. In addition, this case report highlights the need to consider insulin autoantibody syndrome even in patients presenting with fasting hypoglycemic attacks.

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“…First reported in Japan, insulin autoimmune syndrome (IAS) is characterized by the presence of spontaneous hypoglycemia [Okabe et al 1999; Wang et al 2015; Zhang and Zhao, 2014]. High concentrations of total immunoreactive insulin and insulin-binding autoantibodies (IAAs) in IAS lead to severe, recurrent, fasting or late postprandial hypoglycemia [Hakamata et al 1995; Zhang and Zhao, 2014]. Since the initial discovery of IAS, there have been a small number of reported cases predominantly in Japan where IAS is the third leading cause of spontaneous hypoglycemia, following insulinoma and diffuse islet-cell hyperplasia [Hirata et al 1972; Masjhur and Hirata, 1983; Zhang and Zhao, 2014].…”

Section: Introductionmentioning

confidence: 99%