Insulin Autoimmune Syndrome (Hirata Disease): A Rare Cause of Hypoglycemia

Po-Tsang Chen ； Chewn-Yi Yang ； Kai-Jen Tien ； Nai-Cheng Yeh ； Mei-Chen Yeh ； Shang-Yu Lee

《Formosan Journal of Endocrinology and Metabolism》 9卷1期 (2018/06) Pp. 33-37

https://doi.org/10.6856/FJEM.201806_9(1).0005

摘要

Background. Insulin autoimmune syndrome (Hirata disease) is caused by high titer autoimmune antibody binding with endogenous insulin, which can lead to development of recurrent hypoglycemia. Use of certain medications, concomitant autoimmune disease, and specific HLA genotype may increase risk of this syndrome. Methods. We present a case with recurrent hypoglycemia. Insulin autoimmune syndrome was considered the likely diagnosis. Results. A 63-year-old woman had complained of recurrent hypoglycemia for about a year. At admission, we found that she had low plasma glucose levels and extremely high plasma levels of insulin, C-peptide, and insulin antibody. To rule out other causes of endogenous hyperinsulinemic hypoglycemia, abdominal ultrasound and magnetic resonance imaging were performed with negative findings. According to the medical records, the patient took clopidogrel regularly, which led us to suspect drug-induced insulin autoimmune syndrome. Prednisolone therapy was prescribed and clopidogrel was discontinued. We explained to her how to eat a low carbohydrate diet. Soon, her hypoglycemia subsided, and she was discharged from the hospital. Conclusion. Insulin autoimmune syndrome (Hirata disease) is a rare cause of hypoglycemia. Clinicians should be able differentiate it from other causes of endogenous hyperinsulinemia hypoglycemia.

關鍵字

hypoglycemia ； endogenous hyperinsulinism ； insulin ； autoimmunity