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下颌骨戈谢病-斯托特病:一例病例报告及文献综述。

Mandibular Gorham-Stout disease: A case report and literature review.

作者信息

Liu Min, Liu Weiwei, Qiao Chunyan, Han Bing

机构信息

Department of Oral and Maxillofacial Surgery Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling Department of Oral Pathology, School and Hospital of Stomatology, Jilin University, Changchun, China.

出版信息

Medicine (Baltimore). 2017 Oct;96(42):e8184. doi: 10.1097/MD.0000000000008184.

DOI:10.1097/MD.0000000000008184
PMID:29049202
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5662368/
Abstract

RATIONALE

Gorham-Stout disease (GSD) is characterized by aggressive bone resorption, proliferation of vascular or lymphatic vessels, and soft-tissue swelling. Bones that initially appear normal start to resorb, partially or completely. However, the etiology of GSD is unknown.

PATIENT CONCERNS

A 29-year-old man with a chief complaint of toothache and mobility in the lower right mandible for the previous 1 year.

DIAGNOSES

Gorham-Stout disease (GSD).

INTERVENTIONS

The RANK-ligand inhibitor denosumab was suggested to use to inhibit the development of osteoclasts and slow mandibular resorption. In addition, we proposed resection of the remaining mandible and reconstruction via vascularized bone graft, after resorption of the mandible had become stationary.

OUTCOMES

Regular follow-ups were advised to this patient to monitor the stability of bone resorption prior to any surgical intervention.

LESSONS

We strongly recommend that every attempt should be made for early diagnosis and prompt effective medical and surgical management. The failure to do so results in further complications and poor prognosis.

摘要

理论依据

戈勒姆-斯托特病(GSD)的特征是进行性骨吸收、血管或淋巴管增生以及软组织肿胀。最初看似正常的骨骼开始部分或完全吸收。然而,GSD的病因尚不清楚。

患者情况

一名29岁男性,主要诉求是过去1年右下下颌骨牙痛及松动。

诊断

戈勒姆-斯托特病(GSD)。

干预措施

建议使用核因子κB受体活化因子配体抑制剂地诺单抗来抑制破骨细胞的发育并减缓下颌骨吸收。此外,我们建议在颌骨吸收静止后,切除剩余下颌骨并通过带血管蒂骨移植进行重建。

结果

建议该患者定期随访,以在任何手术干预之前监测骨吸收的稳定性。

经验教训

我们强烈建议尽一切努力进行早期诊断并及时进行有效的药物和手术治疗。不这样做会导致进一步的并发症和不良预后。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/9afadbb6bebe/medi-96-e8184-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/bca4f46896cc/medi-96-e8184-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/30fc1e869c25/medi-96-e8184-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/9afadbb6bebe/medi-96-e8184-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/bca4f46896cc/medi-96-e8184-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/30fc1e869c25/medi-96-e8184-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2406/5662368/9afadbb6bebe/medi-96-e8184-g003.jpg

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Distraction osteogenesis for correction of post ankylosis mandibular deformities.
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