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沙利度胺治疗梅尔克森-罗森塔尔综合征的疗效:一例报告

Therapeutic response to thalidomide in Melkersson-Rosenthal syndrome: a case report.

作者信息

Medeiros Manoel, Araujo Maria Ilma, Guimarães Newton Sales, Freitas Luiz Antonio Rodrigues, Silva Tania Maria Correia, Carvalho Edgar Marcelino

机构信息

Immunology Service, Hospital Universitario Federal University of Bahia, Salvador, Brazil.

出版信息

Ann Allergy Asthma Immunol. 2002 Apr;88(4):421-4. doi: 10.1016/s1081-1206(10)62375-7.

DOI:10.1016/s1081-1206(10)62375-7
PMID:11995671
Abstract

BACKGROUND

Melkersson-Rosenthal syndrome (MRS) is a rare disorder of unknown etiology characterized by a triad of symptoms: recurrent orofacial swelling, relapsing facial palsy. and a fissured tongue. A differential diagnosis must be made with other granulomatous diseases, such as sarcoidosis and oral Crohn disease; however, the histologic findings of noncaseating, sarcoidal granulomas support the diagnosis of MRS.

RESULTS

Many therapeutic modalities have been described for this disease. In this case report, we present a patient with MRS that was treated with thalidomide because of the identification of tumor necrosis factor a in the lesion by immunohistochemical analysis. This is the first reported detection of tumor necrosis factor a in an MRS lesion, as well the first reported use of thalidomide to treat this clinical condition.

摘要

背景

梅尔克森 - 罗森塔尔综合征(MRS)是一种病因不明的罕见疾病,其特征为三联征症状:复发性口面部肿胀、复发性面神经麻痹和沟纹舌。必须与其他肉芽肿性疾病进行鉴别诊断,如结节病和口腔克罗恩病;然而,非干酪样、结节样肉芽肿的组织学表现支持MRS的诊断。

结果

针对这种疾病已描述了多种治疗方法。在本病例报告中,我们介绍了一名患有MRS的患者,因其病变通过免疫组织化学分析鉴定出肿瘤坏死因子α而接受了沙利度胺治疗。这是首次报道在MRS病变中检测到肿瘤坏死因子α,也是首次报道使用沙利度胺治疗这种临床病症。

相似文献

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Therapeutic response to thalidomide in Melkersson-Rosenthal syndrome: a case report.沙利度胺治疗梅尔克森-罗森塔尔综合征的疗效:一例报告
Ann Allergy Asthma Immunol. 2002 Apr;88(4):421-4. doi: 10.1016/s1081-1206(10)62375-7.
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The Melkersson-Rosenthal syndrome: a retrospective study of biopsied cases.梅尔基奥森-罗森塔尔综合征:活检病例的回顾性研究。
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Melkersson-Rosenthal syndrome and orofacial granulomatosis.梅尔克森-罗森塔尔综合征与口面部肉芽肿病
Dermatol Clin. 1996 Apr;14(2):371-9. doi: 10.1016/s0733-8635(05)70363-6.
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[The Melkersson-Rosenthal syndrome. A report of 2 cases].[梅尔克森-罗森塔尔综合征。2例报告]
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[Melkersson-Rosenthal syndrome treated by thalidomide. 2 cases].[沙利度胺治疗梅尔克森-罗森塔尔综合征。2例]
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An extraordinary form of the Melkersson-Rosenthal syndrome successfully treated with the tumour necrosis factor-α blocker adalimumab.一种采用肿瘤坏死因子-α阻滞剂阿达木单抗成功治疗的罕见型梅尔克森-罗森塔尔综合征。
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Minerva Stomatol. 1991 Sep;40(9):569-72.

引用本文的文献

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Management Strategies of Melkersson-Rosenthal Syndrome: A Review.梅尔克森-罗森塔尔综合征的管理策略:综述
Int J Gen Med. 2020 Feb 26;13:61-65. doi: 10.2147/IJGM.S186315. eCollection 2020.
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Melkersson-Rosenthal syndrome.梅尔克森-罗森塔尔综合征
J Neurosci Rural Pract. 2014 Nov;5(Suppl 1):S112-4. doi: 10.4103/0976-3147.145258.
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Granulomatous cheilitis: successful treatment of two recalcitrant cases with combination drug therapy.肉芽肿性唇炎:联合药物治疗成功治愈两例顽固性病例
Case Rep Dermatol Med. 2014;2014:509262. doi: 10.1155/2014/509262. Epub 2014 Oct 15.
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An extraordinary form of the Melkersson-Rosenthal syndrome successfully treated with the tumour necrosis factor-α blocker adalimumab.一种采用肿瘤坏死因子-α阻滞剂阿达木单抗成功治疗的罕见型梅尔克森-罗森塔尔综合征。
BMJ Case Rep. 2014 May 14;2014:bcr2014204674. doi: 10.1136/bcr-2014-204674.
5
[Cheilitis granulomatosa Melkersson-Rosenthal syndrome].[唇炎肉芽肿性梅尔克森-罗森塔尔综合征]
Hautarzt. 2006 Feb;57(2):121-6. doi: 10.1007/s00105-005-0981-y.