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증례 : 신장 ; 결절성경화증 환자에서 신장과 간 혈관근지방종 및 폐 침범 2예
이현희 ( Hyun Hee Lee ),정우경 ( Woo Kyung Chung ),장제현 ( Jae Hyun Chang ),정지용 ( Ji Young Jung ),성지윤 ( Ji Yoon Sung ),나선영 ( Sun Young Na ),성연미 ( Yon Mi Sung ) 대한내과학회 2010 대한내과학회지 Vol.79 No.6
결절성경화증은 지능저하, 간질, 피지선종이 특징적으로 나타나는 상염색체 우성 유전 질환으로 뇌, 눈, 심장, 폐, 간, 콩팥, 피부 등 여러 장기에 양성 종양성 병변을 일으키는 질환이다. 혈관근지방종은 혈관, 평활근, 지방조직으로 구성된양성 종양으로, 결절성경화증과 연관되어 발생하는 경우도 많다. 혈관근지방종은 신 실질에 가장 흔하며, 이외 간, 비장, 자궁관, 림프조직 등의 복부 장기 및 폐, 심장에서 발생하기도 한다. 결절성경화증에서 종양이 여러 장기에 동시에 생긴예는 매우 드물다. 저자들은 결절성경화증 환자에서 신장과간에 혈관근지방종이 있고 임파관평활근종증 혹은 다발성 미세결절폐세포증식증 등의 폐 침범을 나타내는 2예를 경험 하였기에 보고하는 바이다. Tuberous sclerosis (TS) involves multiple organs. Angiomyolipoma of the liver or kidney is one of the clinical manifestations of TS. However, coexistent renal and hepatic angiomyolipoma associated with TS is a rare condition. Pulmonary involvement is extremely rare, and occurs in only 0.1~1% of TS. We report two cases of concurrent renal and hepatic angiomyolipomas with pulmonary involvement in patients with TS. The first case was a 35 year-old woman who showed multiple angiomyolipomas in the liver and both kidneys and cystic parenchymal changes in the lungs. The other case was a 27-year-old woman who showed multiple angiomyolipomas in the liver and both kidneys, and multinodular pulmonary shadows. To the best of our knowledge, this is the first reported case of renal and hepatic angiomyolipomas associated with pulmonary involvement in Korea. (Korean J Med 79:705-709, 2010)
전수연 ( Su Yeon Chon ),김유진 ( Yu Jin Kim ),경선영 ( Sun Young Kyung ),안창혁 ( Chang Hyeok An ),이상표 ( Sang Pyo Lee ),박정웅 ( Jeong Woong Park ),정성환 ( Sung Hwan Jeong ),조은경 ( Eun Kyung Cho ),성연미 ( Yon Mi Sung ),김 대한결핵 및 호흡기학회 2010 Tuberculosis and Respiratory Diseases Vol.68 No.2
A 23-year old woman was admitted to our hospital with hemoptysis. The chest X-ray showed reticulonodular opacity and multiple cysts throughout the entire lung field. The chest CT scan revealed numerous bilateral cysts with various sizes, some of them with thickened walls. An open lung wedge resection was performed. The resected specimen showed scattered small nodules, 0.3 to 0.6 cm in size. Microscopically, each nodule was composed of atypical glands with an occasional papillary architecture spreading to the alveolar septa, which were morphologically consistent with a papillary adenocarcinoma with a bronchioloalveolar carcinoma growth pattern. Immunochemically, the tumor cells were negative for the S-100 protein. The patient was diagnosed with an adenocarcinoma of the lung. A variety of diseases can produce or mimic multiple, thin-walled cysts in the lung. Lung cancer with multiple cysts is quite rare. Nevertheless, adenocarcinoma should be a diagnostic consideration. We report a case of a multiple cystic adenocarcinoma of the lung.
기관과 기관지 내 미만성 점막 병변과 좌심실 내 혈전을 동반한 Churg-Strauss 증후군
나선영 ( Sun Young Na ),전수연 ( Soo Yeon Jeon ),김유진 ( Yu Jin Kim ),박정웅 ( Jeong Woong Park ),정성환 ( Sung Hwan Jeong ),이상표 ( Sang Pyo Lee ),성연미 ( Yon Mi Sung ),박상희 ( Sang Hui Park ) 대한천식알레르기학회 2010 천식 및 알레르기 Vol.30 No.4
Churg-Strauss syndrome is a rare syndrome that is characterized by hypereosinophilic systemic necrotizing vasculitis affecting small- to medium-sized arteries and veins. The manifestations of multiple tracheobronchial mucosal lesion and thrombus of left ventricle are rarely reported in Churg-Strauss syndrome. We report a case of Churg-Strauss syndrome with diffuse tracheobronchial mucosal lesions in a 52-year-old man who developed a left ventricular thrombus after discontinuation of treatment. Bronchial asthma, rhinosinusitis, peripheral eosinophilia, p-ANCA positive, eosinophilic lung infiltration, vasculitis led to a diagnosis of Churg-Strauss syndrome. On bronchoscopic evaluation, diffuse necrotizing mucosal lesions were observed on tracheobronchial tree. With methylprednisolone (125 mg/day), his asthmatic symptoms were well-controlled and eosinophil count and eosinophil cationic protein concentration in peripheral blood maintained in normal ranges over a 20 month period, but he was lost to follow-up. After 12 months from discontinuation of treatment he readmitted with relapsed Churg-Strauss syndrome with left ventricular thrombus. The thrombus gradually disappeared after prednisolone (1 mg/kg/day) and warfarin treatment. (Korean J Asthma Allergy Clin Immunol 2010;30:314-319)