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头颈部广泛骨肉性结节病:一种罕见的表现。

Extensive bony sarcoidosis of the head and neck region: a rare presentation.

机构信息

Oral and Maxillofacial Surgery, Chesterfield Royal Hospital, Chesterfield, UK

Oral and Maxillofacial Surgery, Chesterfield Royal Hospital NHS Foundation Trust, Chesterfield, Derbyshire, UK.

出版信息

BMJ Case Rep. 2021 Jan 18;14(1):e237105. doi: 10.1136/bcr-2020-237105.

Abstract

We present a rare case of sarcoidosis with extensive bony destruction of the maxillofacial and skull base bones. A 65-year-old woman was referred with an asymptomatic, non-healing dental socket. Examination revealed an oroantral fistula that was biopsied and repaired under general anaesthesia. Investigations included plain and cross-sectional imaging. Serological tests, in particular ACE, were normal. Histology showed benign florid granulomatous inflammation. At 6 months, the patient remained asymptomatic. She was re-referred 3 years later with further bony destruction of her maxilla and mandible. Repeat imaging showed intrathoracic lymphadenopathy and skull base involvement. Repeat biopsy confirmed granulomatous inflammation. Given the pulmonary, histological and radiological findings, a sarcoidosis diagnosis was made. Following multidisciplinary team meetings, the patient was treated with methotrexate and arrangements made for close monitoring. This case highlights the need for a consensus in identifying, treating and developing a follow-up protocol in such patients.

摘要

我们报告了一例罕见的伴有颌面部和颅底骨广泛骨破坏的结节病。一名 65 岁女性因无症状、不愈合的牙槽窝而被转介。检查发现一个口鼻腔瘘管,在全身麻醉下进行了活检和修复。检查包括平扫和横断面成像。血清学检查,特别是 ACE,正常。组织学显示良性弥漫性肉芽炎症。6 个月后,患者仍无症状。3 年后,她因上颌骨和下颌骨进一步骨破坏而再次被转介。重复成像显示胸腔内淋巴结病和颅底受累。重复活检证实为肉芽肿性炎症。鉴于肺部、组织学和影像学发现,诊断为结节病。经过多学科团队会议,患者接受了甲氨蝶呤治疗,并安排了密切监测。本例强调了在这类患者中需要达成共识,以确定、治疗和制定随访方案。

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本文引用的文献

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Osteolysis of unknown origin: a case report.不明原因的骨质溶解:一例报告
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Osteolytic Sarcoidosis of the Orbital Roof Masquerading as a Malignant Orbital Lesion.伪装成恶性眼眶病变的眶顶溶骨性结节病
Ophthalmic Plast Reconstr Surg. 2017 May/Jun;33(3S Suppl 1):S94-S96. doi: 10.1097/IOP.0000000000000536.
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Sarcoidosis of the head and neck.头颈部结节病
Head Neck Pathol. 2015 Jun;9(2):260-8. doi: 10.1007/s12105-014-0568-y. Epub 2014 Sep 3.
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Osseous sarcoid with lytic lesions in skull.颅骨出现溶骨性病变的骨结节病。
Rheumatol Int. 2014 Apr;34(4):579-82. doi: 10.1007/s00296-013-2752-x. Epub 2013 Apr 23.

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