Long-term efficacy of the sodium-glucose cotransporter 2 inhibitor, ipragliflozin, in a case of type A insulin resistance syndrome.,Journal of Diabetes Investigation

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Long-term efficacy of the sodium-glucose cotransporter 2 inhibitor, ipragliflozin, in a case of type A insulin resistance syndrome.
Journal of Diabetes Investigation ( IF 3.1 ) Pub Date : 2020-03-20 , DOI: 10.1111/jdi.13241
Shuichi Nagashima ₁ , Tetsuji Wakabayashi ₁ , Naoko Saito ₁ , Manabu Takahashi ₁ , Kenta Okada ₁ , Ken Ebihara ₁ , Shun Ishibashi ₁

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Type A insulin resistance (IR) syndrome is a severe IR form caused by insulin receptor (INSR) gene defects. Antidiabetic drugs, including high‐dose insulin and insulin‐sensitizing agents, often fail to control associated hyperglycemia. Therapy with recombinant human insulin‐like growth factor 1 can be more effective, but it is expensive. We report a case of type A IR syndrome with an in‐frame INSR heterozygous deletion (ΔLeu999) that was treated with a combination of conventional therapy and ipragliflozin, a sodium–glucose cotransporter 2 inhibitor. Treatment reduced hemoglobin A1c levels (10.0–7.5%) and induced weight loss (54.4–52.0 kg) within 2 months, and the effects were sustained for >3 years. Sodium–glucose cotransporter 2 inhibitors might be useful to normalize blood glucose in type A IR syndrome by reducing bodyweight and ameliorating glucotoxicity.

中文翻译：

钠-葡萄糖协同转运蛋白 2 抑制剂伊格列净对 A 型胰岛素抵抗综合征的长期疗效。

A 型胰岛素抵抗 (IR) 综合征是由胰岛素受体 ( INSR ) 基因缺陷引起的严重 IR 形式。抗糖尿病药物，包括大剂量胰岛素和胰岛素增敏剂，通常无法控制相关的高血糖。使用重组人胰岛素样生长因子 1 进行治疗可能更有效，但价格昂贵。我们报告了一例具有框内INSR杂合缺失 (ΔLeu999) 的 A 型 IR 综合征病例，该病例采用常规疗法和伊格列净（一种钠-葡萄糖协同转运蛋白 2 抑制剂）联合治疗。治疗在 2 个月内降低了糖化血红蛋白水平 (10.0–7.5%) 并导致体重减轻 (54.4–52.0 kg)，且效果持续超过 3 年。钠-葡萄糖协同转运蛋白 2 抑制剂可能有助于通过减轻体重和改善糖毒性来使 A 型 IR 综合征的血糖正常化。

更新日期：2020-03-20

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