Type B Insulin Resistance in Peru

Zelada, Henry; Gamarra, Dante; Arbañil, Hugo; Manrique, Hélard

doi:10.1016/j.amjms.2016.07.001

Cited by 3 publications

(4 citation statements)

References 18 publications

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“…This review resulted in the selection of 65 articles covering 115 clinical cases of TBIRS, as shown in Figure 1. The articles included were published by (5), hepatitis C (6,7), and human T-cell lymphotropic virus (8). Two cases were associated with Hodgkin's lymphoma (2,9).…”

Section: Resultsmentioning

confidence: 99%

Type B insulin resistance syndrome: a systematic review

Martins

Fernandes

Carvalho

et al. 2020

Archives of Endocrinology and Metabolism

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A literature review on the clinical, laboratory, and treatment features of type B insulin resistance syndrome (TBIRS). Data from PubMed, the Virtual Health Library and Cochrane database were selected and analyzed using the REDCap application and R statistical program. From 182 papers, 65 were selected, which assessed 119 clinical cases, 76.5% in females and 42.9% in African-Americans, with an average age of 44 years. A common feature of TBIRS is co-occurrence of autoimmune diseases, such as systemic lupus erythematosus (most frequently reported). Hyperglycemia of difficult control was the mostly reported condition. Tests for anti-insulin receptor antibodies were positive in 44.2% of the cases. Disease management comprised fractional diet, insulin therapy (maximum dose given was 57 600 IU/day), plasmapheresis and immunosuppression with several classes of drugs, mainly glucocorticoids. Remission occurred in 69.7% of cases, in 30.3% of these spontaneously. The mortality rate was 15.38%. There was an inverse relationship between anti-insulin antibodies and remission (p = 0.033); and a positive correlation between combined immunosuppressive therapy and remission (p = 0.002). Relapse occurred in 7.6% of the cases. This rare syndrome has difficult-to-control diabetes, even with high doses of insulin, and it is usually associated with autoimmune diseases. Therapeutic advances using immunomodulatory therapy have led to significant improvements in the rate of remission.

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Section: Resultsmentioning

confidence: 99%

Type B insulin resistance syndrome: a systematic review

Martins

Fernandes

Carvalho

et al. 2020

Archives of Endocrinology and Metabolism

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“…This is the third case of TBIR to be reported in Peru. The other two cases were middle-aged women, one without active disease, but showing positive markers for Hashimoto's thyroiditis and mixed connective tissue disease, whereas the other woman had rheumatoid arthritis, systemic lupus erythematosus, and Hashimoto's thyroiditis ( 6 ). It should be noted that most patients do not me et al the criteria for a specific autoimmune disease, but may have one or more autoimmune features, such as alopecia, nephritis, anaemia, leukopenia, and positive antinuclear antibodies ( 12 ).…”

Section: Discussionmentioning

confidence: 99%

“…In one case, betamethasone was administered at low doses for 30 months ( 15 ). Two other cases were reported in Peru: in the first case, 100 mg of cyclophosphamide was given orally for 3 weeks; and in the second case, eight pulses of 1 g of cyclophosphamide every 28 days in combination with prednisone 20 mg/day were administrated, with remission occurring 1 year later ( 6 ). In the present case, the patient received six pulses of methylprednisolone and 6 months of cyclophosphamide treatment, and achieved remission, defined as euglycaemia without the use of insulin and normalisation of fasting insulin concentrations ( Table 2 ).…”

Section: Discussionmentioning

confidence: 99%

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Successful remission of type B insulin resistance syndrome without rituximab in an elderly male

Concepción-Zavaleta

Ildefonso-Najarro

Dueñas

et al. 2020

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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Summary Type B insulin resistance syndrome (TBIR) is a rare autoimmune disease caused by antibodies against the insulin receptor. It should be considered in patients with dysglycaemia and severe insulin resistance when other more common causes have been ruled out. We report a case of a 72-year-old male with a 4-year history of type 2 diabetes who presented with hypercatabolism, vitiligo, acanthosis nigricans, and hyperglycaemia resistant to massive doses of insulin (up to 1000 U/day). Detection of anti-insulin receptor antibodies confirmed TBIR. The patient received six pulses of methylprednisolone and daily treatment with cyclophosphamide for 6 months. Response to treatment was evident after the fourth pulse of methylprednisolone, as indicated by weight gain, decreased glycosylated haemoglobin and decreased requirement of exogenous insulin that was later discontinued due to episodes of hypoglycaemia. Remission was eventually achieved and the patient is currently asymptomatic, does not require insulin therapy, has normal glycaemia and is awaiting initiation of maintenance therapy with azathioprine. Thus, TBIR remitted without the use of rituximab. This case highlights the importance of diagnosis and treatment in a timely fashion, as well as the significance of clinical features, available laboratory findings and medication. Large controlled studies are required to standardise a therapeutic protocol, particularly in resource-constrained settings where access to rituximab is limited. Learning points: Type B insulin resistance syndrome is a rare autoimmune disorder that should be considered in patients with dysglycaemia, severe insulin resistance and a concomitant autoimmune disease. Serological confirmation of antibodies against the insulin receptor is not necessary in all cases due to the high associated mortality without timely treatment. Although there is no standardised immunosuppressive treatment, a protocol containing rituximab, cyclophosphamide and steroids has shown a significant reduction in previously reported mortality rates. The present case, reports successful remission in an atypical patient using cyclophosphamide and methylprednisolone, which is an effective therapy in countries in which rituximab is not covered by health insurance. When there is improvement in the hypercatabolic phase, the insulin dose should be reduced and/or discontinued to prevent hypoglycaemia; a mild postprandial hyperglycaemic state should be acceptable.

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