Tuberculosis and Respiratory Diseases

The Korean Academy of Tuberculosis and Respiratory Diseases (대한결핵및호흡기학회)
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A Case of Hughes-Stovin Syndrome

Hughes-Stovin Syndrome 1예

  • Kim, Joo-In (Department of Internal Medicine, Inje University College of Medicine) ;
  • Lee, Young-Min (Department of Internal Medicine, Inje University College of Medicine) ;
  • Yum, Ho-Kee (Department of Internal Medicine, Inje University College of Medicine) ;
  • Choi, Soo-Jeon (Department of Internal Medicine, Inje University College of Medicine) ;
  • Choi, Seok-Jin (Department of Radiology, Inje University College of Medicine) ;
  • Yang, Young-Il (Department of Pathology, Inje University College of Medicine) ;
  • Sung, Kyu-Bo (Department of Radiology, Asan Medical Center, Ulsan University) ;
  • Kim, Dong-Soon (Department of Internal Medicine, Asan Medical Center, Ulsan University) ;
  • Lee, Bong-Choon (Department of Internal Medicine, Inje University College of Medicine)
  • 김주인 (인제대학교 의과대학 내과학교실) ;
  • 이영민 (인제대학교 의과대학 내과학교실) ;
  • 염호기 (인제대학교 의과대학 내과학교실) ;
  • 최수전 (인제대학교 의과대학 내과학교실) ;
  • 최석진 (인제대학교 의과대학 진단방사선학교실) ;
  • 양영일 (인제대학교 의과대학 병리학교실) ;
  • 성규보 (울산대학교 의과대학 진단방사선학교실) ;
  • 김동순 (울산대학교 의과대학 내과학교실) ;
  • 이봉춘 (인제대학교 의과대학 내과학교실)
  • Published : 1996.02.29
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Abstract

Hughes-Stovin Syndrome is an exceedingly rare combination of distal pulmonary arterial aneurysm and deep vein thrombosis, mostly found in young patients. There are striking similarities between the vascular manifestation of Behçet's disease and Hughes-Stovin Syndrome. It has been suggested that they may have a similar pathogenesis. Most patients died of massive hemoptysis due to rupture of aneurysm. Recently we have experienced the first case of Hughes-Stovin Syndrome in Korea. A 37 year old male patient was admitted because of recurrent hemoptysis and intermittent fever. He had a history of recurrent aphthous ulcers and erythema nodosum-like skin rash, But no other findings of Behcet's disease was found. Angiography showed multiple pulmonary arterial aneurysm and deep vein thrombosis in Right lower extremity. Histologic examination of specimens of open lung biopsy revealed leukocytoclastic angiitis. Pulmonary arterial aneurysms were successfully treated by coil embolization and he is in good condition with corticosteroid and cyclophosphamide therapy.

저자들은 과거력상 반복되는 구강내 아프타성 궤양 및 결절홍반양 피부병변이 있었고, 객혈을 주소로 내원한 37세 남자환자에서 혈관조영술상 다발성 폐동맥류, 심부 정맥의 협착 및 폐색소견이 관찰되었고, 조직학적으로는 폐혈관염 소견을 보여, Hughes-Stovin 증후군으로 진단하였으며, 코일을 이용한 폐동맥색전술로 폐동맥류를 성공적으로 치료하였기에 문헌고찰과 함께 보고하는 바이다.

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