Rituximab for the treatment of type B insulin resistance syndrome: a case report and review of the literature
Rituximab for the treatment of type B insulin resistance syndrome: a case report and review of the literature
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DOI：
10.1111/dme.13524
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发表时间：
2017-12-01
影响因子：
3.5
通讯作者：
Shibata, T.
中科院分区：
文献类型：
--
作者：
Iseri, K.;Iyoda, M.;Shibata, T.
背景：B型胰岛素抵抗综合征是一种罕见的疾病，其特征是难治性的一过性高血糖和与循环中的抗胰岛素受体抗体相关的严重的胰岛素抵抗。B型胰岛素抵抗综合征的标准化治疗方案尚未建立。病例报告我们报告一例，男性，因抗中性粒细胞胞浆抗体相关性小血管炎和糖尿病肾病而接受血液透析，并迅速出现高血糖(糖化白蛋白52.1%)。根据抗胰岛素受体抗体阳性和男性抗中性粒细胞胞浆抗体相关小血管炎和特发性血小板减少性紫癜的自身免疫史，诊断为B型胰岛素抵抗综合征。尽管糖皮质激素和大剂量胰岛素治疗后仍有严重的高血糖，但利妥昔单抗治疗能显著改善患者的严重胰岛素抵抗和抗胰岛素受体抗体的消失，且无任何不良反应。结论根据对11例患者的文献复习，利妥昔单抗是一种安全有效的治疗激素抵抗的B型胰岛素抵抗综合征的策略。
Background Type B insulin resistance syndrome is a rare disease characterized by refractory transient hyperglycaemia and severe insulin resistance associated with circulating anti-insulin receptor antibodies. A standardized treatment regimen for type B insulin resistance syndrome has yet to be established.Case report We report the case of a 64-year-old man undergoing haemodialysis for antineutrophil cytoplasmic antibody-associated vasculitis and diabetic nephropathy, who developed rapid onset of hyperglycaemia (glycated albumin 52.1%). Type B insulin resistance syndrome was diagnosed, on the basis of positivity for anti-insulin receptor antibodies and the man's autoimmune history of antineutrophil cytoplasmic antibody-associated vasculitis and idiopathic thrombocytopenic purpura. Although severe hyperglycaemia persisted in spite of corticosteroids and high-dose insulin therapy, rituximab treatment resulted in remarkable improvement of the man's severe insulin resistance and disappearance of anti-insulin receptor antibodies without any adverse effects.Conclusions According to a literature review of 11 cases in addition to the present case, rituximab appears to be a safe and effective strategy for the treatment of corticosteroid-resistant type B insulin resistance syndrome.