하악골에 발생한 Gorham-Stout 증후군: 증례 보고 = GORHAM-STOUT DISEASE OF THE MANDIBLE: A CASE REPORT

국문 초록 (Abstract)

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.

다국어 초록 (Multilingual 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.

참고문헌 (Reference)

1 Moller G, "The Gorham-Stout syndrome (Gorham’s massive osteolysis). A report of six cases with histopathological findings" 81 : 501-, 1999

2 Heffez L, "Perspectives on massive osteolysis. report of a case and review of the literature" 55 : 331-, 1983

3 Johnson PM, "Observations on massive osteolysis; a review of the literature and report of a case" 71 : 28-, 1958

4 Branco F, "Notes on a rare case of essential osteolysis" 40 : 519-, 1958

5 Ohya T, "Massive osteolysis of the maxillofacial bones. report of two cases" 70 : 698-, 1990

6 Benhalima H, "Massive osteolysis of the maxillo-facial bones: case report and review of the literature" 24 : 35-, 2001

7 Booth DF, "Massive osteolysis of the mandible: an attempt at reconstruction" 32 : 787-, 1974

8 Mendez AA, "Massive osteolysis of the femur (Gorham’s disease): a case report and review of the literature" 9 : 604-, 1989

9 Gorham LW, "Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis" 37 : 985-, 1955

10 Tsang WM, "Massive osteolysis (Gorham disease) of the maxillofacial skeleton: report of 2 cases" 62 : 225-, 2004

11 Devlin RD, "Interleukin-6: a potential mediator of the massive osteolysis in patients with Gorham-Stout disease" 81 : 1893-, 1996

12 Dunbar SF, "Gorham’s massive osteolysis: the role of radiation therapy and a review of the literature" 26 : 491-, 1993

13 Dominguez R, "Gorham’s disease or vanishing bone disease: plain film, CT, and MRI findings of two cases" 24 : 316-, 1994

14 Spieth ME, "Gorham’s disease of the radius: radiographic, scintigraphic, and MRI findings with pathologic correlation. a case report and review of the literature" 26 : 659-, 1997

15 Anavi Y, "Gorham’s disease affecting the maxillofacial skeleton" 11 : 550-, 1989

16 Florchinger A, "Gorham-Stout syndrome of the spine. Case report and review of the literature" 168 : 68-, 1998

17 Bode-Lesniewska B, "Gorham-Stout disease of the shoulder girdle and cervicothoracic spine: fatal course in a 65-year-old woman" 31 : 724-, 2002

18 Ceroni D, "Gorham-Stout disease of costo-vertebral localization: radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings" 45 : 464-, 2004

19 Chung C, "Gorham syndrome of the thorax and cervical spine: CT and MRI findings" 26 : 55-, 1997

20 Chong Ng L, "Gorham disease of the cervical spinea case report and review of the literature" 28 : 355-, 2003

21 Manisali M, "Gorham disease : correlation of MR findings with histopathologic changes" 8 : 1647-, 1998

22 Hirayama T, "Cellular and humoral mechanisms of osteoclast formation and bone resorption in Gorham-Stout disease" 195 : 624-, 2001

23 Vinee P, "CT and MRI of Gorham syndrome" 18 : 985-, 1994

24 Jackson JBS, "A boneless arm" 18 : 368-, 1838