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혼합형 자가면역성용혈성빈혈과 동반된 Evans 증후군 1례
선길홍,윤찬영,박상곤,박경희,우정주,한경택,김진화,김영훈,정춘해,박치영 朝鮮大學校 附設 醫學硏究所 2005 The Medical Journal of Chosun University Vol.30 No.2
Evans syndrome is defined as a simultaneous or sequential occurrence of autoimmune hemolytic anemia and autoimmune thrombocytopenia. Autoimmune hemolytic anemia (AIHA) is usually induced by IgG warm autoantibody or cold-active IgM antibodies reacting specifically with antigens associated with a patient's RBC. AIHA is a fairly uncommon disorder, with estimates of the incidence at 1 to 3 cases per 100,000 per year. Mixed-type AIHA is a relatively uncommon form of AIHA, with studies noting the incidence of 7-8% among cases of AIHA. We experienced a patient, 46-year-old woman who was diagnosed having a very rare clinical presentation of mixed warm and cold antibody mediated Evans syndrome. She was treated with corticosteroid therapy only and has been maintaining a complete response for 15 weeks. 저자들은 혼합형의 자가면역성 용혈성 빈혈과 자가면역성 혈소판 감소증이 동시에 발생한 혼합형 Evans 증후군으로 진단하고 스테로이드 요법 후 혈액학적으로 회복된 상태로 15주가 지난 현재 steroid 5 mg/일 까지 감량한 상태에서 추적 관찰 치료중인 46세 여자 환자를 경험하였기에 문헌 고찰과 함께 보고하는 바이다.
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선길홍,윤찬영,정춘해,박치영 朝鮮大學校 附設 醫學硏究所 2006 The Medical Journal of Chosun University Vol.31 No.3
Background: Small cell carcinomas (SCC) are most commonly seen in the lung, approximately 2.5-4% of all SCC occur in extrapulmonary sites. This study aimed to review the clinical features, therapy and natural course of patients with extrapulmonary small cell carcinoma (EPSCC) in single-institution series. Methods: We retrospectively reviewed the medical records of patients with SCC between December 2002 and November 2005. Study eligibility required that patients had pathologically proven SCC in sites other than lung with normal radiological findings of the chest and normal sputum cytology or negative bronchoscopic findings. Result: Eight patients with EPSCC were identified and their primary sites were various such as esophagus (4 cases), kidney (1 case), thymus (1 case), stomach (1 case) and unknown primary supraclavicular lymph node (1 case). The 4 patients were classified as limited disease (LD) and 2 patients were as extended disease (ED) at initial staging work-up. But 2 patients were undetermined. The 3 patients with EPSCC of LD were treated with operation and adjuvant chemotherapy or chemotherapy and radiotherapy and one received only supportive care. They showed favorable clinical courses with a median overall survival of 23 months. EPSCC of ED and undetermined group received only supportive care. They had aggressive clinical courses with a median overall survival of 3 months, Conclusions: EPSCC was identified in various sites with the most common primary site being the esophagus, It was predominantly occurred in old male patients and associated with smoking like as SCC of the lung. Since EPSCC is a relalively rare disease that mimics SCC of the lung in its response to treatment and survival pattern, it would appear advisable to follow similar treatment guidelines. Although recurrence within short terms was observed, a significant survival benefit was obtained in EPSCC of LD received treatment (chemotherapy, operation and radiation therapy). Thus, EPSCC is usually a fatal disease with meidan overall survival of 18 months. 연구배경: 소세포암의 가장 흔한 발생부위는 폐이며, 폐외소세포암은 전체 소세포암의 2.5~4%를 차지하는 매우 드문질환이다. 본 연구의 목적은 단일 기관에서 진단된 폐외소세포암의 임상적 특징, 치료와 자연경과를 보고하고자 하였다. 방법: 조선대학교부속병원에서 2002년 12월 부터 2005년 11월 사이에 소세포암으로 진단된 66명의 의무기록을 검토하였다. 폐외소세포암은 흉부 단순 몇 전산화단층 촬영, 객담세포 검사, 기관지경 검사에서 폐병번이 없으면서 폐와 병변의 조직학적 생검에서 소세포암으로 증명된 경우로 하였으며, 총 8명이였다. 결과: 본 연구에서 식도가 전체 폐외소세포 암 환자의 50%(4례)로 가장 많은 원발병소를 보였으며 그 외 흉선, 신장, 위가 1례씩 있었으며 쇄곧상부 림프절에서 소세포암이 발견되었으나 원발 병소를 발견 못한 경우가 1례 관찰되었다. 4명의 환자는 제한 병기를 보였으며 2명은 광범위 병기를 보였지만 나머지 2명은 위내시경으로 식도에서 소세포암이 조직학적으로 진단되었으나 보전적 치료만을 받길 원하여 병기결정을 위한 검사는 시행할 수 없었다. 제한 병기의 폐외소세포암 환자는 4명이었다. 2명은 수술적 절제후 보조항암화학요법을 받았으며, 1명은 항암화학요법후 방사션치료를 받았고, 나머지 한명은 보전적 치료만 시행하였다. 제한 병기의 폐외소세포암 환자틀은 양호한 임상 경과를 보였으며 중앙 생존기간은 23개월이었다. 하지만 광범위 병기의 폐외소세포암 환자와 병기를 분류할 수 없던 환자에 대해서는 항암화학요법 등의 적극적 치료는 시행하지 않고 보존적 치료만 시행하였다. 그들의 임상경과는 빠르게 악화되었으며 중앙 생존 기간은 3개월이었다. 결론: 본 연구에서 폐외소세포암은 다양한 장기에서 발견될 수 있으나 가장 흔한 곳은 식도였다. 소세포폐암과 유사하게 고령, 남성 및 흡연과 관련을 보였었다. 제한 병기에서 국소 치료, 항암화학요법은 생존기간의 연장을 보였으나 쉽게 다른 장기로 전이가 되거나 재발하는 매우 치명적인 질환임을 확인할 수 있었다. 전체 중앙 생존기간은 18개월 이었고 항암 화학요법등의 치료를 받은 군이 보존적 치료를 받은 군에 비해 의미있는 생존기간 (p=0.040)의 연장을 보였다. 본 연구는 단일기관의 상대적으로 적은 수의 소규모 연구이지만 폐외소세포암의 제한 병기에서 국소치료 및 항암화학요법은 생존기간의 연장을 보였고 쉽게 다른 장기로 전이가 되거나 재발하는 매우 치명적인 질환임을 확인 할 수 있었다.
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폐흡충에 의한 무균성 노흉에 대한 돼지꼬리형 도관(pig-tail catheter)을 이용한 치료의 효과
한경택,권세훈,김형호,하재화,선길홍,권용은,윤성호,이승일 朝鮮大學校 附設 醫學硏究所 2006 The Medical Journal of Chosun University Vol.31 No.3
Background: Pulmonary paragonimiasis is caused by consumption of raw or improperlycooked crustacea infected with the laval stage (metacercaria) of Paragonimus westermani. The most characteristic symptoms were rust-colored sputum and cough. Paragonimiasis causes pleural thickening or effusion in 48% of the patients. Pleuro-pulmonary paragonimiasis can be easily overlooked by physicians who do not suspect this disease in the differential diagnosis. Method: We compared the outcomes of 11 patients with paragonimus empyema managed either through thoracotomy or pig-tail catheter drain. These patients were confirmed by food history, clinical and radiological findings, and laboratory data. Results: The male and female ratio was 1.75 : 1, and mean age was 40.0 ± 13.5 years. AII patients had pulmonary symptoms such as cough or chest pain. Serum-ELISA for paragonimiasis were all positive (mean titer was 0.57). AII patients had pleural effusion in radiological findings( 2 patients had bilateral pleural effusion). All patients received praziquantel (75 ㎎/㎏/day for 3days). Two patients were treated with thoracotomy and nine patients were treated with pig-tail drain. Hospital stay were 14.5 days in thoracotomy group and 5.6 days in pig-tail group respectively, Conclusion: Compared to the conventional thoracotomy grouP, the patients with paragonimus empyema who received pig-tail catheters had a significantly-decreased period of drain in situ, were clinically improved earlier, and were discharged earlier.
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SLC25A13 유전자분석으로 확인된 성인형 제II형 Citrullinemia 1예
김진화,박거운,박철진,선길홍,김형호,김상용,배학연,조은해,양송현 대한내분비학회 2006 Endocrinology and metabolism Vol.21 No.6
Adult onset type II citrullinemia is an autosomal recessive disorder of the amino acid metabolism caused by a deficiency of liver specific argininosuccinate synthetase activity. This disease can occur at any age in life with recurrent episodes of neurological signs and symptoms such as disorientation, abnormal behaviors (aggression, irritability and hyperactivity), seizures, coma and potential death from brain edema, which are resulted from hyperammonemia. We should consider this rare metabolic disease for the adult patient who exhibits mental change and hyperammonemia without liver or brain disease. Recently. SLC25A13 gene, encoding the mitochondrial aspartate glutamate carrier protein named citrin, is demonstrated to be responsible for adult onset type II citrullinemia. We experienced a 39-year-old female who suffered from generalized weakness, dizziness and lethargy, and diagnosed as adult onset type II citrullinemia by highly elevated plasma citrulline and ammonia and the SLC25A13 gene mutation. We report here on this unusual case of adult onset type II citrullulinemia with a brief review of the related literature. (J Kor Endocrinol Soc 21:542~547, 2006) 의식변화가 동반된 지속되는 고암모니아혈증을 주소로 내원하여 SLC25A13 유전자 분석에 의해 돌연변이가 확인된 성인형 제II형 citrullinemia 1예를 경험하였기에 보고하는 바이다.
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