Insulin-induced autoimmune syndrome: A case report

Chu, Jianping; Zheng, Xiaowei; Liu, Jie; Zhong, Jinyan; Li, Jialin; Xu, Miao; Fan, Lin

doi:10.3892/etm.2016.3767

Cited by 15 publications

(15 citation statements)

References 14 publications

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“…Despite this clinical difference, the only reliable method for the differential diagnosis between these two forms of autoimmune hypoglycemia is the characterization of the autoantibodies: as a matter of fact, IAA are present in IAS, whereas anti-receptor insulin antibodies are present in type B insulin resistance. 112…”

Section: Diagnosismentioning

confidence: 99%

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

Cappellani¹,

Macchia²,

Falorni³

et al. 2020

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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: Diagnosismentioning

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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“…Management of IAS as reported by di erent case reports included using di erent immunosuppressive regimens, such as prednisolone [12], hydrocortisone [13], cyclophosphamide [14], azathioprine [15], rituximab [16], mycophenolate mofetil [17], and plasmapheresis [18,19]. Our patient had a complete recovery a er 8-weeks course of prednisolone, although many case reports documented a recovery period a er 12-weeks of prednisolone [20,21]. IAS could be a misleading condition and careful diagnosis is needed to rule out other causes of hyperinsulinaemic hypoglycaemia, especially in patients with previous history of bariatric surgery or signs of insulinoma.…”

Section: Discussionmentioning

confidence: 71%

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

Alrashidi

Alessa²

2019

Case Reports in Endocrinology

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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. e patient revealed that he had taken several di erent 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. e patient had elevated fasting insulin level (>301 uIU/mL) and elevated insulin autoantibodies (>100.0 IU/mL). A er appropriate work-up, he was diagnosed with IAS. A er treatment with prednisolone (1 mg/kg/day), the patient had complete recovery. In patients with repeated hypoglycaemia, IAS should be considered in the di erential diagnosis. Glucocorticoid therapy can be e ective for the treatment of hypoglycaemia in patients with IAS.

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“…It is caused by the agonistic effect of antibodies against the insulin receptor resulting in significant insulin resistance and paradoxical hypoglycemia. In these cases, anti-receptor insulin antibodies are usually positive whereas anti-insulin antibodies are negative (4,15). Therefore, considering that our patient did not present acanthosis nigricans, and laboratory results showed high levels of insulin and anti-insulin antibodies, IAS seems to be the most likely diagnosis.…”

Section: Discussionmentioning

confidence: 72%

“…Alpha-glucosidase inhibitors have also been used to reduce or prevent hypoglycemic episodes (11,12). In more severe or prolonged cases, glucocorticoids, immunosuppressants, and even plasmapheresis may be useful as adjuvant therapy (3,15). Other reported alternatives include pancreatectomy, diazoxide, and octreotide (15).…”

Section: Discussionmentioning

confidence: 99%

“…In more severe or prolonged cases, glucocorticoids, immunosuppressants, and even plasmapheresis may be useful as adjuvant therapy (3,15). Other reported alternatives include pancreatectomy, diazoxide, and octreotide (15). Immunoadsorption using a reusable adsorber system loaded with sheep antigens directed against human immunoglobulin followed by two doses of 1 g of rituximab has been reported effective in a patient with IAS refractory to prednisolone and azathioprine therapy (19).…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

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

Reis¹,

Fernandes²,

Fontenele³

et al. 2018

Archives of Endocrinology and Metabolism

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Insulin autoimmune syndrome (IAS, Hirata's disease) is a rare hypoglycemic disorder characterized by spontaneous hypoglycemia associated with extremely high circulating insulin levels and positive anti-insulin antibody results. Thus far, most cases have been reported in Asian countries, notably Japan, with few cases reported in western countries. As a possible cause, it is associated with the use of drugs containing sulfhydryl radicals, such as captopril. This report refers to a 63-year-old female Brazilian patient with a history of postprandial hypoglycemia. After extensive investigation and exclusion of other causes, her hyperinsulinemic hypoglycemia was considered to have likely been induced by captopril. Most cases of IAS are self-limiting. However, dietary management, corticosteroids, plasmapheresis, and rituximab have already been used to treat patients with IAS. In our case, after discontinuation of captopril, an initial decrease in insulin autoantibody levels was observed followed by improvement in episodes of hypoglycemia. Although it is a rare disease, IAS should be considered in the differential diagnosis of endogenous hyperinsulinemic hypoglycemia. Patients with suspected IAS must be screened for autoimmunity-related drugs for insulin. Initial clinical suspicion of IAS can avoid unnecessary costs associated with imaging examinations and/or invasive surgical procedures.

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Insulin-induced autoimmune syndrome: A case report

Cited by 15 publications

References 14 publications

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

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

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

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

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