Zeng Xiang Xia, Tang Yun Liang, Hu Kai Xiang, Wang Jiao, Zhu Ling Yan, Liu Jian Ying, Xu Jixiong
Department of Endocrinology and Metabolism, First Affiliated Hospital of Nanchang University, Nanchang, Jiangxi, China.
Medicine (Baltimore). 2017 Dec;96(51):e9213. doi: 10.1097/MD.0000000000009213.
Insulin autoimmune syndrome (IAS) is an uncommon disorder characterized by hyperinsulinemic hypoglycemia related to insulin-binding autoantibodies. To the best of our knowledge, we report the first case of a pregnant female with IAS.
The 26-year-old patient with Graves disease and 10 weeks pregnant developed IAS after approximately 6 months treatment with methimazole. The patient exhibited recurrent spontaneous hypoglycemia.
On evaluation, laboratory findings detected both high fasting insulin (>1000 mIU/L) and insulin autoantibodies. An oral glucose tolerance test showed elevated insulin concentrations with disproportionately elevated C-peptide levels. The imaging study showed nomasslesionsinthepancreas,and the patient was clinically diagnosed with IAS.
The patient had an abortion, discontinued methimazole and switched to oral prednisone (30 mg once daily) and propylth- iouracil (100 mg 3 times daily) for 3 months.
At the 3-month follow-up visit, hypoglycemic episodes had disappeared and insulin antibody levels were no longer detectable.
We have described this case and reviewed the relevant literature concerning diagnosis and treatment of IAS. Importantly, this case indicates that clinicians should view pregnancy as another factor of hypoglycemia in IAS.
胰岛素自身免疫综合征(IAS)是一种罕见的疾病,其特征为与胰岛素结合自身抗体相关的高胰岛素血症性低血糖症。据我们所知,我们报告了首例患有IAS的怀孕女性病例。
这位26岁患有格雷夫斯病且怀孕10周的患者在接受甲巯咪唑治疗约6个月后患上了IAS。患者出现反复自发性低血糖症。
经评估,实验室检查发现空腹胰岛素水平高(>1000 mIU/L)且存在胰岛素自身抗体。口服葡萄糖耐量试验显示胰岛素浓度升高,同时C肽水平不成比例地升高。影像学检查显示胰腺无占位性病变,该患者临床诊断为IAS。
患者进行了人工流产,停用甲巯咪唑,改用口服泼尼松(每日一次,30 mg)和丙硫氧嘧啶(每日三次,100 mg),持续3个月。
在3个月的随访中,低血糖发作消失,胰岛素抗体水平不再可检测到。
我们描述了该病例并回顾了有关IAS诊断和治疗的相关文献。重要的是,该病例表明临床医生应将怀孕视为IAS中低血糖的另一个因素。