国际口腔医学杂志

国际口腔医学杂志 ›› 2018, Vol. 45 ›› Issue (4): 480-484.doi: 10.7518/gjkq.2018.04.019

• 综述 • 上一篇    下一篇

口腔颌面部溶骨症

刘敏, 刘炜炜, 徐志民, 王梓霖, 李佳乐, 韩冰   

  1. 吉林大学口腔医院口腔颌面外科,吉林省牙发育及颌骨重塑与再生重点实验室 长春 130021
  • 收稿日期:2017-09-22 修回日期:2018-02-17 出版日期:2018-07-02 发布日期:2018-07-02
  • 通讯作者: 韩冰,教授,博士,Email：hbing@jlu.edu.cn
  • 作者简介:刘敏,住院医师,硕士,Email：1522212026@qq.com
  • 基金资助:
    国家自然科学基金（81602377）

Gorham-Stout syndrome in the maxillofacial region

Liu Min, Liu Weiwei, Xu Zhimin, Wang Zilin, Li Jiale, Han Bing.   

  1. Dept. of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University;
    Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, China
  • Received:2017-09-22 Revised:2018-02-17 Online:2018-07-02 Published:2018-07-02
  • Supported by:
    This study was supported by National Natural Science Foundation of China (81602377).

RichHTML

11

PDF (PC)

253

摘要： 溶骨症又称Gorham-Stout综合征,以自发性进行性骨吸收为主要特点,在临床上较罕见,其病因及发病机制尚不明确。病理以薄壁血管增生和纤维组织代替消失的骨组织为主要特征,临床表现多样,可发生于身体多处骨组织,甚或侵及临近的软组织。目前国内外关于颌面部溶骨症的报道较少,而且治疗方法的效果评价没有明确的标准。本文根据国内外文献,对该病的病因、临床表现、诊断与鉴别诊断及治疗进行简要综述。

关键词: Gorham-Stout综合征, 溶骨症, 颌面骨

Abstract: Gorham-Stout syndrome, which is also called massive osteolysis, vanishing bone disease, phantom bone disease, is characterized by spontaneous progressie bone resorption, and is a rare disorder in the clinical, whereas the etiology is unknown. Gorham-Stout syndrome is characterized histologically by the proliferation of endothelial-lined vessels in bone and by the replacement of bone with fibrous tissue. Clinical manifestations are diverse. In most cases, only a single bone or several bones are affected. Associated infiltrative soft tissue abnormality is often found. Reports of maxillofacial bone resorption are few. According to the literature of domestic and overseas, the aim of this paper is to make a brief review about the etiology, clinical manifestations, diagnosis and differential diagnosis and therapy of Gorham-Stout syndrome.

Key words: Gorham-Stout syndrome, massive osteolysis, maxillofacial bone

中图分类号: 

  • R782
[1] Gorham LW, Stout AP. Massive osteolysis(acute spontaneous absorption of bone, phantom bone, disa-ppearing bone); its relation to hemangiomatosis[J]. J Bone Joint Surg Am,1955, 37-A(5): 985-1004.
[2] Dellinger MT, Garg N, Olsen BR.Viewpoints on vessels and vanishing bones in Gorham-Stout disease[J]. Bone, 2014, 63: 47-52.
[3] Patel DV.Gorham’s disease or massive osteolysis[J]. Clin Med Res, 2005, 3(2): 65-74.
[4] Shi J, Zhang Z, Li Y, et al.Reossification in Gorham’s disease of the hand and wrist with unusual CT and MR imaging features[J]. Skeletal Radiol, 2015, 44(7): 1033-1038.
[5] Yalniz E, Alicioglu B, Benlier E, et al.Gorham-Stout disease of the humerus[J]. JBR-BTR, 2008, 91(1): 14-17.
[6] 游云华, 汪跃平, 梁军, 等. 上颌骨溶骨症一例报告并文献复习[J]. 中华肿瘤防治杂志, 2010, 17(14): 1131-1133.You YH, Wang YP, Liang J, et al.Massive osteo-lysis of maxillary bone: report of one case and litera-ture review[J]. Chin J Cancer Prev Treat, 2010, 17(14): 1131-1133.
[7] 李运良, 刘京兵, 朱兆夫. 颅、颌骨溶解症2例报道[J]. 口腔颌面外科杂志, 2002, 12(2): 183-184.Li YL, Liu JB, Zhu ZF.Report of 2 cases of jaw osteolysis[J]. J Oral Maxillofac Surg, 2002, 12(2): 183-184.
[8] Escande C, Schouman T, Françoise G, et al.Histo-logical features and management of a mandibular Gorham disease: a case report and review of maxil-lofacial cases in the literature[J]. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 2008, 106(3): e30-e37.
[9] Perschbacher SE, Perschbacher KA, Pharoah MJ, et al.Gorham’s disease of the maxilla: a case report[J]. Dentomaxillofac Radiol, 2010, 39(2): 119-123.
[10] Maillot C, Cloche T, Le Huec JC.Thoracic osteotomy for Gorham-Stout disease of the spine: a case report and literature review[J]. Eur Spine J, 2014. [Epub ahead of print].
[11] Hardegger F, Simpson LA, Segmueller G.The syn-drome of idiopathic osteolysis. Classification, review, and case report[J]. J Bone Joint Surg Br, 1985, 67(1): 88-93.
[12] Busilacchi A, Ramazzotti D, Ulisse S, et al.Gorham-Stout disease as a complication of posterior shoulder capsulorrhaphy[J]. J Shoulder Elbow Surg, 2012, 21(9): e1-e7.
[13] Devlin RD, Bone HG 3rd, Roodman GD. Interleukin-6: a potential mediator of the massive osteolysis in pa-tients with Gorham-Stout disease[J]. J Clin Endo-crinol Metab, 1996, 81(5): 1893-1897.
[14] Hirayama T, Sabokbar A, Itonaga I, et al.Cellular and humoral mechanisms of osteoclast formation and bone resorption in Gorham-Stout disease[J]. J Pathol, 2001, 195(5): 624-630.
[15] Spieth ME, Greenspan A, Forrester DM, et al.Gor-ham’s disease of the radius: radiographic, scintigraphic, and MRI findings with pathologic correlation. A case report and review of the literature[J]. Skeletal Radiol, 1997, 26(11): 659-663.
[16] Venkatramani R, Ma NS, Pitukcheewanont P, et al.Gorham’s disease and diffuse lymphangiomatosis in children and adolescents[J]. Pediatr Blood Cancer, 2011, 56(4): 667-670.
[17] Dupond JL, Bermont L, Runge M, et al.Plasma VEGF determination in disseminated lymphangiomatosis-Gorham-Stout syndrome: a marker of activity? A case report with a 5-year follow-up[J]. Bone, 2010, 46(3): 873-876.
[18] Zhu X, Gao JJ, Landao-Bassonga E, et al.Thonzonium bromide inhibits RANKL-induced osteoclast forma-tion and bone resorption in vitro and prevents LPS-induced bone loss in vivo[J]. Biochem Pharmacol, 2016, 104: 118-130.
[19] Eghbali-Fatourechi G, Khosla S, Sanyal A, et al.Role of RANK ligand in mediating increased bone resorption in early postmenopausal women[J]. J Clin Invest, 2003, 111(8): 1221-1230.
[20] Kong YY, Feige U, Sarosi I, et al.Activated T cells regulate bone loss and joint destruction in adjuvant arthritis through osteoprotegerin ligand[J]. Nature, 1999, 402(6759): 304-309.
[21] Darnay BG, Ni J, Moore PA, et al.Activation of NF-kappaB by RANK requires tumor necrosis factor receptor-associated factor (TRAF) 6 and NF-kappa B-inducing kinase. Identification of a novel TRAF6 interaction motif[J]. J Biol Chem, 1999, 274(12): 7724-7731.
[22] Armstrong AP, Tometsko ME, Glaccum M, et al.A RANK/TRAF6-dependent signal transduction path-way is essential for osteoclast cytoskeletal organiza-tion and resorptive function[J]. J Biol Chem, 2002, 277(46): 44347-44356.
[23] Feng X.RANKing intracellular signaling in osteo-clasts[J]. IUBMB Life, 2005, 57(6): 389-395.
[24] Kai B, Ryan A, Munk PL, et al.Gorham disease of bone: three cases and review of radiological features[J]. Clin Radiol, 2006, 61(12): 1058-1064.
[25] Zhao SF, Wang YX, Yang XD, et al.Gorham disease of the mandible[J]. J Craniofac Surg, 2015, 26(2): e160-e162.
[26] Yang RH, Chen PC, Chang CP, et al.Gorham’s disease: the disappearing bone[J]. Clin Nucl Med, 2013, 38(5): 361-364.
[27] Lala S, Mulliken JB, Alomari AI, et al.Gorham-Stout disease and generalized lymphatic anomaly—clinical, radiologic, and histologic differentiation[J]. Skeletal Radiol, 2013, 42(7): 917-924.
[28] Alves VM, Vieira TS, Amorim NS, et al.99mTc(V)-DMSA SPECT-CT findings in a case of Gorham-Stout disease[J]. Nucl Med Rev Cent East Eur, 2015, 18(2): 97-101.
[29] Liu Y, Zhong DR, Zhou PR, et al.Gorham-Stout disease: radiological, histological, and clinical fea-tures of 12 cases and review of literature[J]. Clin Rheumatol, 2016, 35(3): 813-823.
[30] Suero Molina EJ, Niederstadt T, Ruland V, et al.Cerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine[J]. J Neuro-surg Spine, 2014, 21(6): 956-960.
[31] Choi BS, Hong SJ, Chu MA, et al.Gastrointestinal tract involvement of Gorham’s disease with expre-ssion of D2-40 in duodenum[J]. Pediatr Gastroen-terol Hepatol Nutr, 2014, 17(1): 52-56.
[32] Franchi A, Bertoni F, Bacchini P, et al.CD105/endo-glin expression in Gorham disease of bone[J]. J Clin Pathol, 2009, 62(2): 163-167.
[33] Kuriyama DK, McElligott SC, Glaser DW, et al. Treatment of Gorham-Stout disease with zoledronic acid and interferon-α: a case report and literature review[J]. J Pediatr Hematol Oncol, 2010, 32(8): 579-584.
[34] Edwards JR, Williams K, Kindblom LG, et al.Lym-phatics and bone[J]. Hum Pathol, 2008, 39(1): 49-55.
[35] Chattopadhyay P, Bandyopadhyay A, Das S, et al.Gorham’s disease with spontaneous recovery[J]. Singapore Med J, 2009, 50(7): e259-e263.
[36] Shimizu T, Sato K, Yoshida T, et al.A case report of Gorham-Stout syndrome remission[J]. J Orthop Sci, 2012, 17(2): 199-204.
[37] Lala S, Mulliken JB, Alomari AI, et al.Gorham-Stout disease and generalized lymphatic anomaly—clinical, radiologic, and histologic differentiation[J]. Skeletal Radiol, 2013, 42(7): 917-924.
[38] Ruggieri P, Montalti M, Angelini A, et al.Gorham-Stout disease: the experience of the Rizzoli Institute and review of the literature[J]. Skeletal Radiol, 2011, 40(11): 1391-1397.
[39] Aizawa T, Sato T, Kokubun S.Gorham disease of the spine: a case report and treatment strategies for this enigmatic bone disease[J]. Tohoku J Exp Med, 2005, 205(2): 187-196.
[40] Paley MD, Lloyd CJ, Penfold CN.Total mandibular reconstruction for massive osteolysis of the mandible (Gorham-Stout syndrome)[J]. Br J Oral Maxillofac Surg, 2005, 43(2): 166-168.
[41] Sekharappa V, Arockiaraj J, Amritanand R, et al.Gorham’s disease of spine[J]. Asian Spine J, 2013, 7(3): 242-247.
[42] Lehmann G, Pfeil A, Böttcher J, et al.Benefit of a 17-year long-term bisphosphonate therapy in a pa-tient with Gorham-Stout syndrome[J]. Arch Orthop Trauma Surg, 2009, 129(7): 967-972.
[43] Möller G, Priemel M, Amling M, et al.The Gorham-Stout syndrome (Gorham’s massive osteolysis). A report of six cases with histopathological findings[J]. J Bone Joint Surg Br, 1999, 81(3): 501-506.
[44] Kuriyama DK, McElligott SC, Glaser DW, et al. Treatment of Gorham-Stout disease with zoledronic acid and interferon-α: a case report and literature review[J]. J Pediatr Hematol Oncol, 2010, 32(8): 579-584.
[45] Grunewald TG, Damke L, Maschan M, et al.First report of effective and feasible treatment of multi-focal lymphangiomatosis (Gorham-Stout) with beva-cizumab in a child[J]. Ann Oncol, 2010, 21(8): 1733-1734.
[46] Takahashi A, Ogawa C, Kanazawa T, et al.Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome[J]. J Pediatr Surg, 2005, 40(3): E47-E50.
[47] Dupond JL, Bermont L, Runge M, et al.Plasma VEGF determination in disseminated lymphangio-matosis-Gorham-Stout syndrome: a marker of activity? A case report with a 5-year follow-up[J]. Bone, 2010, 46(3): 873-876.
[48] 黄慧, 徐作军, 王立, 等. Gorham-Stout综合征五例临床分析并文献复习[J]. 中华内科杂志, 2009, 48(1): 23-27.Huang H, Xu ZJ, Wang L, et al.Clinical features of Gorham-Stout syndrome and literature review[J]. Chin J Int Med, 2009, 48(1): 23-27.
[49] Heyd R, Micke O, Surholt C, et al.Radiation therapy for Gorham-Stout syndrome: results of a national patterns-of-care study and literature review[J]. Int J Radiat Oncol Biol Phys, 2011, 81(3): e179-e185.
[1] 石海涛,黄金霞,潘剑. 内镜技术在上颌窦异物取出术中的应用进展[J]. 国际口腔医学杂志, 2020, 47(4): 452-457.
[2] 吴勇志,尧可,陈思宇,黄能文,王了. 多生牙及其诊断治疗[J]. 国际口腔医学杂志, 2020, 47(3): 311-317.
[3] 马心笛,陈蕾. 完全脱位牙再植的牙髓、牙周膜愈合：从生物学基础到牙外伤指南[J]. 国际口腔医学杂志, 2020, 47(3): 336-344.
[4] 陈艺尹,刘俊圻,李承浩. 牙槽突裂的裂隙特点及正畸治疗对唇腭裂患者牙槽突植骨术的影响[J]. 国际口腔医学杂志, 2020, 47(3): 345-350.
[5] 谢瑜,周懿. 截冠术：下颌第三磨牙保守拔除术[J]. 国际口腔医学杂志, 2020, 47(1): 17-21.
[6] 王家云,胡欢,林云红. 数字化改良式与传统基托式塞治器在下颌骨开窗减压术中的对比研究[J]. 国际口腔医学杂志, 2020, 47(1): 37-42.
[7] 林阳阳,侯敏. 双侧下颌支矢状骨劈开术对下颌近心骨段位移变化的影响[J]. 国际口腔医学杂志, 2019, 46(6): 718-723.
[8] 张晓敏,高莺. 残余颌下腺Wharton导管内涎石复发的研究进展[J]. 国际口腔医学杂志, 2019, 46(5): 604-608.
[9] 邓程丹,石冰,李杨. 唇腭裂患者的脑部结构与功能研究进展[J]. 国际口腔医学杂志, 2019, 46(5): 617-620.
[10] 李承浩,李业平,石冰. 唇裂鼻畸形整复核心问题之华西观点[J]. 国际口腔医学杂志, 2019, 46(4): 383-386.
[11] 李业平,郑谦,尹恒,郭春丽,石冰,李承浩. 影响单侧唇裂鼻畸形二期整复主观评价结果的鼻测量指标探究[J]. 国际口腔医学杂志, 2019, 46(4): 407-412.
[12] 刘小旭,马坚,石冰,黄永清. Pai综合征的研究进展[J]. 国际口腔医学杂志, 2019, 46(3): 339-342.
[13] 刘丹,毛渤淳,雒如燕,崔珂,石冰,龚彩霞. 唇腭裂患者家庭抗逆力及其影响因素[J]. 国际口腔医学杂志, 2019, 46(3): 297-301.
[14] 闫冰,骆献阳,谭迎赟,关丽梅,薛丽丽. 血清表面增强拉曼光谱应用于口腔鳞状细胞癌患者临床分期的研究[J]. 国际口腔医学杂志, 2019, 46(3): 277-281.
[15] 何映酉,乎森,李继华. 外科辅助快速上颌扩张的临床研究进展[J]. 国际口腔医学杂志, 2019, 46(3): 343-348.
Viewed
Full text


Abstract

Cited
  Shared   
  Discussed   
[1] 张新春. 桩冠修复与无髓牙的保护[J]. 国际口腔医学杂志, 1999, 26(06): .
[2] 王昆润. 长期单侧鼻呼吸对头颅发育有不利影响[J]. 国际口腔医学杂志, 1999, 26(05): .
[3] 彭国光. 颈淋巴清扫术中颈交感神经干的解剖变异[J]. 国际口腔医学杂志, 1999, 26(05): .
[4] 杨凯. 淋巴化疗的药物运载系统及其应用现状[J]. 国际口腔医学杂志, 1999, 26(05): .
[5] 康非吾. 种植义齿下部结构生物力学研究进展[J]. 国际口腔医学杂志, 1999, 26(05): .
[6] 柴枫. 可摘局部义齿用Co-Cr合金的激光焊接[J]. 国际口腔医学杂志, 1999, 26(04): .
[7] 孟姝,吴亚菲,杨禾. 伴放线放线杆菌产生的细胞致死膨胀毒素及其与牙周病的关系[J]. 国际口腔医学杂志, 2005, 32(06): 458 -460 .
[8] 费晓露,丁一,徐屹. 牙周可疑致病菌对口腔黏膜上皮的粘附和侵入[J]. 国际口腔医学杂志, 2005, 32(06): 452 -454 .
[9] 庞莉苹,姚江武. 抛光和上釉对陶瓷表面粗糙度、挠曲强度及磨损性能的影响[J]. 国际口腔医学杂志, 2008, 35(S1): .
[10] 李子夏,包广洁. 龋病病因相关因素的激光扫描共聚焦显微镜研究[J]. 国际口腔医学杂志, 2008, 35(S1): .