Continuous glucose monitoring and Rituximab treatment in insulin autoimmune syndrome

Boro, Hiya; Gupta, Uttio; Singh, Charandeep; Malhotra, Rakhi; Khadgawat, Rajesh

doi:10.1016/j.dsx.2021.102294

Cited by 3 publications

(5 citation statements)

References 16 publications

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“…The use of continuous glucose monitoring systems has also demonstrated the effectiveness of plasma exchange in treating insulin autoimmune symptoms by normalizing blood glucose and antibody levels, although it is not a curative treatment. The use of continuous glucose monitoring systems provides a way to precisely assess the efficacy of different drugs [ 56 ].…”

Section: Resultsmentioning

confidence: 99%

“…At present, the treatment plan for insulin syndrome has not been unified, and various treatment plans have been proposed in various studies, including drug withdrawal, frequent small meals, avoidance of monosaccharide intake, glucocorticoid treatment [ 59 , 60 ], plasma exchange [ 61 ], immunosuppressive agents (such as sulfur azathioprine [ 56 ] and 6-mercaptopurine), and rituximab [ 56 , 61 , 62 ] ( Table 6 ).…”

Section: Resultsmentioning

confidence: 99%

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Insulin Autoimmune Syndrome: A Systematic Review

Lin

Chen

Ning

2023

International Journal of Endocrinology

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Insulin autoimmune syndrome (IAS) is a rare endocrine disorder characterized by recurrent episodes of severe hypoglycemia, markedly elevated serum insulin, and positive insulin autoantibodies. In recent years, various countries have reported it one after another. It can be seen that we must pay attention to this disease. The diagnosis of IAS is challenging, requiring a careful workup aimed at excluding other causes of hyperinsulinemic hypoglycemia. High levels of insulin autoantibodies are found in patients, and C-peptide is not parallel to insulin, which could be diagnostic. IAS is a self-limiting disease with a good prognosis. Its treatment mainly includes symptomatic supportive treatment, such as adjusting the diet and using acarbose and other drugs to delay the absorption of glucose to prevent hypoglycemia. For patients with severe symptoms, available treatments may include drugs that reduce pancreatic insulin secretion (such as somatostatin and diazoxide), immunosuppressants (glucocorticoids, zaprin, and rituximab), and even plasma exchange to remove autoantibodies from the body. This review provides a comprehensive analysis of the epidemiology, pathogenesis, clinical manifestations, diagnosis and identification, and monitoring and treatment management of IAS.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Insulin Autoimmune Syndrome: A Systematic Review

Lin

Chen

Ning

2023

International Journal of Endocrinology

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“…When the binding capacity is exceeded, the insulin dissociates from IAA and now causes significant post-prandial hypoglycemia two to five hours after a meal [ 9 , 10 ]. CGM studies have shown IAS to be associated with alternating post-prandial hyperglycemia followed by hypoglycemia [ 7 , 11 ]. Symptoms are initially adrenergic/autonomic (tremors, anxiety, hunger, and sweating), followed by neuroglycopenia (irritability, behavior change, confusion, loss of consciousness, or seizures).…”

Section: Discussionmentioning

confidence: 99%

“…Tapering doses of glucocorticoids have been successfully used to help with the remission or control of hypoglycemia. Other immunosuppressants, like azathioprine [ 17 ] and rituximab [ 7 ], have been used in various case reports with good success. Other treatments like octreotide [ 9 ], diazoxide [ 18 ], and partial pancreatectomy have been less commonly used.…”

Section: Discussionmentioning

confidence: 99%

“…Due to its rare occurrence in Caucasians and the lack of IAA assays in some labs, IAS could often go undiagnosed for a significant period of time, with several unnecessary lab and imaging tests, resulting in significant morbidity for the patients. IAS is mostly a self-remitting disease but can be challenging if it becomes persistent, with treatment being stopping the offending drugs, lowcarbohydrate meal plan, glucocorticoids, alpha-glucosidase inhibitors, immunosuppressant or even plasma exchange [3][4][5]7,8]. There is no clear consensus on treatment guidelines, except for case reports or review papers.…”

Section: Introductionmentioning

confidence: 99%

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Persistent Insulin Autoimmune Syndrome in a Caucasian Male in the Absence of Triggers

Edem,

Maradana,

Majety

et al. 2024

Cureus

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Insulin autoimmune syndrome (IAS) or Hirata disease is a rare condition presenting as recurrent hypoglycemia, and associated with elevated insulin levels in the presence of insulin autoantibodies (IAAs) in patients who were never exposed to exogenous insulin and with no evidence of pancreatic abnormalities. IAS is much more frequent in East Asians, especially the Japanese population, compared to the lower incidence in Caucasians. However, it can be associated with other autoimmune diseases or drug use like methimazole and alpha-lipoic acid (ALA). We report a case of a 47-year-old Caucasian male presenting with a 12-month history of worsening episodes of fasting and post-prandial hypoglycemia associated with symptoms of dizziness, tremors, palpitations, and unconsciousness associated with hypoglycemia. Symptoms resolved with the administration of carbohydrate-containing foods, establishing Whipple’s triad. At an outside facility, he had initial labs that showed elevated insulin levels (141 µU/ml) with normal glucose, C-peptide, and proinsulin levels, but there was no availability of an IAA lab assay. Given his symptoms, severity, and frequency of hypoglycemia, he was admitted to the hospital for a 72-hour fast, which showed the lowest glucose level of 64 mg/dl with inappropriately high insulin of 22.2 µU/ml, low C-peptide of 0.57 ng/ml, and undetectable proinsulin of <1.6 pmol/L, but with IAA being >50 U/ml (0.0-0.4 U/ml). He was treated with intensive dietary counseling with a low-carbohydrate diet and prednisone 20 mg twice daily initially. Additionally, he could not tolerate octreotide, diazoxide, and acarbose due to side effects. He is currently on prednisone 10 mg daily and nifedipine with no further hypoglycemic episodes, but still has a high IAA of >50 U/ml and serum insulin levels of 70-112 µU/ml. Our case highlights the importance of recognizing hypoglycemia and checking for IAA levels as first-line diagnostic tests, in the absence of which there could be a delay in diagnosis and leading to unnecessary lab and imaging testing. Our case is unique since it happened in a Caucasian without any prior exposure to a triggering factor and has not undergone self-remission yet, which happens in most of IAS cases.

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Insulin autoimmune syndrome induced by omeprazole in an Asian Male with HLA-DRB1*0406 Subtype: A case report

Ai,

Zhang,

2024

Diabetes Research and Clinical Practice

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Continuous glucose monitoring and Rituximab treatment in insulin autoimmune syndrome

Cited by 3 publications

References 16 publications

Insulin Autoimmune Syndrome: A Systematic Review

Insulin Autoimmune Syndrome: A Systematic Review

Persistent Insulin Autoimmune Syndrome in a Caucasian Male in the Absence of Triggers

Insulin autoimmune syndrome induced by omeprazole in an Asian Male with HLA-DRB1*0406 Subtype: A case report

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