Int J Stomatol

Int J Stomatol ›› 2023, Vol. 50 ›› Issue (4): 445-451.doi: 10.7518/gjkq.2023051

• Case Report • Previous Articles     Next Articles

Gorham-Stout disease in maxillofacial bone: a case report and literature review

Wu Yifan(),Lu Hao,Liu Shengwen,Xu Wanlin(),Yang Wenjun.   

  1. Dept. of Oral and Maxillofacial-Head and Neck Oncology, Shanghai Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, College of Stomatology, Shanghai Jiao Tong University, National Center for Stomatology, National Clinical Research Center for Oral Diseases, Shanghai Key Laboratory of Stomatology, Shanghai 200011, China
  • Received:2022-11-29 Revised:2023-04-06 Online:2023-07-01 Published:2023-06-21
  • Contact: Wanlin Xu E-mail:Asher0713@163.com;xwlcontribution@163.com
  • Supported by:
    Shanghai Sailing Program(21YF1423500);the Biobank Project of Shanghai Ninth People’s Hospital(YBKB202214)

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Abstract:

Gorham-Stout disease (GSD), also known as massive osteolysis or vanishing bone disease (VBD), is a spontaneous, progressive disease of bone resorption characterized by lymphoid tissue and vascular hyperplasia. GSD does not differ in age, sex, or race and can involve single or multiple bones, but rarely occurs in the maxillofacial region. However, its cause remains controversial, and there is still no unified standard for the evaluation of treatment options and efficacy. In this paper, we report a case of GSD with recurrence in the Department of Oral and Maxillofacial-Head and Neck Onco-logy, Shanghai Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine. In addition, we briefly review the etiology, clinicopathological features, diagnostic points, and treatment of GSD to further enhance our understan-ding of GSD that occurs in the maxillofacial bone.

Key words: Gorham-Stout disease, massive osteolysis, vanishing bone disease, maxillofacial bone

CLC Number: 

  • R 782

TrendMD: 

Fig 1

Preoperative imaging photos"

Fig 2

Histological HE staining for vascular proliferation optical microscope × 100"

Fig 3

Postoperative follow-up panoramic X-ray"

Tab 1

Literature review of GSD in maxillofacial region"

时间/年参考文献

年龄/岁

（性别）

部位组织学表现治疗随访时间/预后
2015[12]29（女）下颌骨致密纤维组织，血管左下颌骨节段性切除术6个月/无进展
2015[13]18（男）下颌骨致密胶原纤维组织手术+二期修复2.5年/无进展
2016[14]15（女）下颌骨、上颌骨、颧骨血管淋巴样增生，炎细胞浸润化学治疗（顺铂+5-氟尿嘧啶）10年/无进展
2016[15]

7（男）

8（男）

39（男）

颅颌面多骨

颞骨

颅颌面多骨

———
2016[16]6（女）下颌骨浆细胞淋巴细胞浸润观察2年/无进展
2016[17]54（女）上颌骨———
2017[18]14（女）下颌骨血管瘤样血管增生——
2017[19]8（女）下颌骨血管内皮细胞增生观察—
2017[20]29（男）下颌骨血管瘤样血管增生地诺单抗—
2017[21]7（女）下颌骨纤维胶原，淋巴细胞浸润，血管增生放射治疗（共计40 Gy）9个月/无进展
2017[22]38（男）下颌骨—阿仑膦酸—
2017[23]13（男）下颌骨纤维组织及血管增生帕米膦酸二钠1年/进展
2017[24]38（男）下颌骨致密纤维组织及血管观察—
2018[25]

6（男），6（男）

9（女），7（男）

下颌骨，颞下颌关节窝—其中3例行双膦酸盐治疗无进展
2018[26]25（男）下颌骨血管瘤样病变阿仑膦酸钠（每周70 mg）14个月/进展
2018[27]51（男）颞骨、颞下颌关节—观察—
2018[28]20~51/2例（男），2例（女）2例下颌骨，1例颌骨和颞骨，1例下颌骨、颞骨和颧骨—阿仑膦酸钠（每周70 mg）—/无进展
2018[29]14（男）下颌骨—干扰素和类固醇6个月/进展
2019[30]34（男），16（男）下颌骨—下颌骨节段性切除术+游离腓骨瓣重建—/进展
2020[31]3（男）下颌骨骨吸收伴淋巴细胞浸润干扰素、唑来膦酸盐、雷帕霉素4年/进展
2020[32]61（女）下颌骨致密结缔组织及扩张血管增生下颌骨病损切除术4个月/无进展
2021[3]60（男）上颌骨慢性炎细胞浸润加巴喷丁6个月/无进展
2021[33]—（女）下颌骨胶原纤维、炎细胞、血管增生——
2022[34]3（女）颞骨炎细胞浸润、血管扩张增生——
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