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증례 : 두 종류의 항체를 보유한 자가면역성 저혈당 1예
전성완 ( Sung Wan Chun ),이병완 ( Byung Wan Lee ),강은석 ( Eun Seok Kang ),차봉수 ( Bong Soo Cha ),이은직 ( Eun Jig Lee ),임승길 ( Sung Kil Lim ),이현철 ( Hyun Chul Lee ) 대한당뇨병학회 2009 임상당뇨병 Vol.10 No.2
자가항체로 인한 고인슐린성 저혈당증을 특징으로 하는 자가면역 저혈당은 인슐린 자가면역 증후군과 B형 인슐린저항성으로 나뉘며, 서로 다른 임상적 특성을 보인다. 저자들은 glucocorticoid 치료에 반응하지 않는 자가면역성 저혈당증에 대해 두 가지 종류의 인슐린 항체가 함께 존재함을 확인하고 cyclophosphamide 충격요법으로 저혈당이 호전된 73세 여자 환자 증례를 경험하였기에 문헌고찰과 함께 보고하는 바이다. Autoimmune hypoglycemia is rare kind of autoimmune disease caused by either anti-insulin antibodies (insulin autoimmune syndrome, IAS) or anti-insulin receptor antibodies (type B insulin resistance, type-B IR). We experienced an extremely rare case of the IAS accompanied by type-B IR. A 73-year-old woman presented with recurrent severe hypoglycemic symptoms at dawn for one month was admitted to the Severance hospital. She had several medical histories including 30 years of hypertension, 15 years of type 2 diabetes, and 4 years of coronary artery disease before admission. She had never received an insulin injection. Hypoglycemia was diagnosed at 10 hours in the 72-hour fasting test with glucose, insulin, and C-peptide levels of 42 mg/dL, 280.31 μU/mL, and 7.70 ng/mL, respectively. The insulin autoantibody titer was 130 μU/mL and quantitative assay for insulin receptor antibody was positive. Insulinoma was ruled out by imaging techniques and calcium stimulation test. She has no evidence of other diseases associated with altered immunity. Despite of treatment with prednisolone, symptomatic hypoglycemic events persisted at fasting state. Early induction of 300 mg cyclophosphamide therapy resulted in remission of hypoglycemia accompanied by suppressed antibody titer. The changes in autoantibodies might result in alleviation of the symptoms of hypoglycemia and improvement in insulin and C-peptide levels. (Korean Clinical Diabetes J 10:123-128, 2009)
면역 글로블린 투여로 호전된 용형성 요독 증후군 1 예
조덕신(Duck Shin Cho),이병재(Byung Jae Lee),민태훈(Tae Hoon Min),강보현(Bo Hyun Kang),김형훈(Hyung Hoon Kim),이병완(Byung Wan Lee),최동철(Dong Chul Choi) 대한내과학회 2002 대한내과학회지 Vol.63 No.4
The hemolytic uremic syndrome (HUS) is a clinical syndrome defined by the presence of thrombocytopenia, microangiopathic hemolytic anemia and acute renal failure with or without a clinically apparent etiology. The conventional treatment of choice is plasmapheresis as a first-line therapy. Most patients respond to plasmapheresis whereas some patients are refractory to the therapy. The second-line therapy is not well established although various therapies such as steroid, vincristine, intravenous immunoglobulin have been suggested. The intravenous immunoglobulin therapy in refractory hemolytic-uremic syndrome have rarely been successful in complete remission. We report a case of refractory HUS in a 48 year-old man who developed hemolytic anemia, thrombocytopenia, acute renal failure and ischemic retinopathy. The patient was refractory to plasmapheresis as a first-line therapy. The patient received intravenous immunoglobulin therapy as a second-line therapy after 8 days of plasmapheresis, which subsequently resulted in a complete remission of refractory HUS. The complete remission using immunoglobulin in HUS has not been previously reported in Korea.(Korean J Med 63:431-435, 2002)