Endocrinology Unit, Azienda Ospedaliera-Universitaria di Padova, Department of Medicine (DIMED), Padova, Italy.
Blood Transfusion Center, Azienda Ospedaliera - Universitaria di Padova, Padova, Italy.
Clin Chem Lab Med. 2018 May 24;56(6):889-895. doi: 10.1515/cclm-2017-0392.
We describe the case of a 54-year-old Caucasian Italian male experiencing episodes of hypoglycemia, occurring mainly after meals. He had never been exposed to insulin and was taking ramipril, flecainide and acetylsalicylic acid. An oral glucose tolerance test (OGTT) showed high blood glucose levels diagnostic for diabetes mellitus at 120 min and hypoglycemia with inappropriately high insulin levels at 240 min. The 72-h fasting test, abdominal computed tomography (CT) and positron emission tomography-CT were normal. Insulin autoantibodies were positive at high titers, prompting a diagnosis of insulin autoimmune syndrome (IAS). The patient was advised to take frequent, small meals and thus achieved a good control of his hypoglycemic symptoms. After 18 months of this dietary management, his insulin autoantibody levels decreased considerably but remained detectable. During an OGTT, his blood glucose levels at 120 min were now indicative of an impaired glucose tolerance rather than diabetes, and there was improvement in the glucose nadir. The patient had no other clinical or latent autoimmune diseases. Here we discuss the main features of IAS (also known as Hirata's disease) and review the cases of IAS reported in Italy to date.
我们描述了一位 54 岁的白人意大利男性的病例，他主要在饭后出现低血糖发作。他从未接触过胰岛素，正在服用雷米普利、氟卡尼和乙酰水杨酸。口服葡萄糖耐量试验（OGTT）显示，120 分钟时血糖水平高，诊断为糖尿病，240 分钟时出现低血糖，胰岛素水平不适当升高。72 小时禁食试验、腹部计算机断层扫描（CT）和正电子发射断层扫描-CT 正常。胰岛素自身抗体呈高滴度阳性，提示胰岛素自身免疫综合征（IAS）的诊断。建议患者少食多餐，从而很好地控制了低血糖症状。在这种饮食管理 18 个月后，他的胰岛素自身抗体水平显著下降，但仍可检测到。在 OGTT 中，他 120 分钟时的血糖水平现在表明糖耐量受损而不是糖尿病，血糖最低点有所改善。患者没有其他临床或潜伏自身免疫性疾病。在这里，我们讨论了 IAS（也称为 Hirata 病）的主要特征，并回顾了迄今为止在意大利报告的 IAS 病例。
