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Rehabilitation Practice and Science

Translated Title

出血性腦中風病人合併胰島素自身免疫綜合症：罕見案例報告與文獻回顧

Abstract

Introduction: Insulin Autoimmune Syndrome (IAS), also known as Hirata's disease, is a rare cause of hyperinsulinemia hypoglycemia, with an overall incidence of one over one billion. Only three hundred and eightycases of IAS were reported world-wide during 1970 to 2009. Such disease was easily underestimated and misdiagnosed. Thus, we sincerely introduce a case newly diagnosed as IAS possibly induced by Angiotensin-Converting Enzyme inhibitor (ACEI) after cerebral vascular accident (CVA).The literature of insulin autoimmune syndrome was also reviewarticleed. Case presentation: This 34-year-old male with past history of hypertension and hyperlipidemia hadsudden onset of mental drowsiness and weakness on left side limbs and was brought to emergent department (ED) for treatment on Oct 8, 2013. As Brain computed tomography (CT) showed right posterior basal ganglion hemorrhage, burrhole hematoma aspiration and intracranial pressure (ICP) monitor were performed. The postoperative course was smooth, and rehabilitation program was arranged afterward due to persistent left limbs weakness and poor walking balance. However, during admission for rehabilitation, multiple episodes of cold sweating and hypoglycemia were recorded. Even though the patient denied a history of type 1 ortype 2 diabetes mellitus and his HbA1c was normal, insulin autoimmune syndrome was suspected. The profile of autoimmune including C3, C4, rheumatic profile, dsDNA, ssDNA, C-peptide, thyroid profile (TSH, T3, free T4), insulin antibody, anti-B2 glycoprotein, anti-cardiolipin, IgG, IgG4, anti-SSa/SSb, ESR, and saliva production tests were checked. Additionally, evidence of other autoimmune diseases such as systemic lupus erythematosus, and hematological disease such as multiple myeloma, were not observed. Furthermore, the data of immune antibody showed elevated insulin antibody (82.1%). Thus, insulin autoimmune syndrome was diagnosed. The treatment of prednisolone 30mg/day was prescribed and gradually titrated to 45mg/day. As the trend of blood sugar became much more smoothly, and the patient was discharged afterward. Conclusions: Insulin autoimmune syndrome is a rare disease that causes hypoglycemia. The pathophysiology of IAS may be due to the high binding capacity and low affinity of insulin antibody(IAA), IAA can binding insulin and spontaneously dissociatelater, which inappropriately increases unbound insulin, resulting in hypoglycemic episodes. In this case, the possible cause of insulin autoimmune syndrome was induced by using captopril in previous medical treatment. Therefore, we suggest that the patient with IAS should avoid using captopril as anti-hypertensive drugs and taking medications may developing IAS which have been reported in the current literatures.

Language

Traditional Chinese

First Page

59

Last Page

65

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