Maxillofacial Plastic and Reconstructive Surgery

Korean Association of Maxillofacial Plastic and Reconstructive Surgeons (대한악안면성형재건외과학회)
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GORHAM-STOUT DISEASE OF THE MANDIBLE: A CASE REPORT

하악골에 발생한 Gorham-Stout 증후군: 증례 보고

  • Ji, Young-Min (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Song, Gin-Ah (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Shin, Jung-Hyun (Research Institute and Hospital, National Cancer Center) ;
  • Pang, Kang-Mi (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Kim, Soung-Min (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Myoung, Hoon (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Seo, Byoung-Moo (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Choi, Jin-Young (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Lee, Jong-Ho (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Choung, Pill-Hoon (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Kim, Myung-Jin (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University) ;
  • Hwang, Soon-Jung (Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University)
  • 지영민 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 송진아 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 신정현 (국립암센터 구강종양클리닉) ;
  • 방강미 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 김성민 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 명훈 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 서병무 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 최진영 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 이종호 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 정필훈 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 김명진 (서울대학교 치과대학 구강악안면외과학교실) ;
  • 황순정 (서울대학교 치과대학 구강악안면외과학교실)
  • Published : 2008.11.30
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Abstract

Gorham-Stout disease is a rare skeletal disease which is progressive and spontaneous and idiopathic resorption of whole body bone. In the initial stage, the osteolytic site is replaced by histologically benign vascular proliferation without new bone formation and finally by dense fibrous tissues. We encountered with a patient involving the mandible with attention by the clinical, radiographic, scintigraphic, angiographic, and computed tomography. We resected the mandibular lesion which was reconstructed with a fibular free flap. We report literature review with a mandibular lesion involved with Gorham-Stout disease.

Gorham-Stout 증후군은 조직학적으로 양성의 혈관 증식을 보이면서 광범위한 골 용해 현상을 나타내는 희귀한 병으로 알려져 있다. 병인론은 아직 명확히 밝혀져 있지 않으며 주로 남성과 젊은 성인에게서 호발하는 것으로 알려져 있다. 진단에는 컴퓨터 단층촬영, 자기공명영상, 핵의학검사, 혈관조영검사 등의 영상검사가 도움이 되며, 조직학적검사 역시 몇몇 특이 소견을 통해 진단에 도움을 줄 수 있다. 발생하는 부위에 따라서 그 예후는 매우 다르며, 특히 척추나 흉곽에 발생할 경우는 생명을 위협할 수 있다. 저자등은 우측 하악골의 병적 골절을 주소로 내원한 환자에서 임상검사, 방사선학적 검사, 조직병리학적 검사를 통해 Gorham-Stout 증후군으로 진단한 환자를 비골유리피판을 이용한 하악골 재건술을 통해 치료하였고 4년 동안 관찰 기간 중 재발 없이 양호한 치유 경과를 보였기에 문헌고찰과 함께 보고하는 바이다.

Keywords

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