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口腔浆细胞性肉芽肿:是免疫球蛋白G4相关性疾病的一种黏膜表现还是一种模仿病变?

Plasma cell granuloma of the oral cavity: a mucosal manifestation of immunoglobulin G4-related disease or a mimic?

作者信息

Laco Jan, Kamarádová Kateřina, Mottl Radovan, Mottlová Alena, Doležalová Helena, Tuček Luboš, Žatečková Kamila, Slezák Radovan, Ryška Aleš

机构信息

The Fingerland Department of Pathology, Charles University in Prague-Faculty of Medicine and University Hospital in Hradec Kralove, Sokolska 581, 500 05, Hradec Kralove, Czech Republic,

出版信息

Virchows Arch. 2015 Mar;466(3):255-63. doi: 10.1007/s00428-014-1711-6. Epub 2014 Dec 19.

DOI:10.1007/s00428-014-1711-6
PMID:25522952
Abstract

The aim of the study was to test the hypothesis that oral plasma cell granuloma may represent a mucosal manifestation of immunoglobulin (Ig)G4-related disease (IgG4-RD) in the oral cavity. The study sample comprised two males and four females, aged 54-79 years (median 62 years). The lesions were localized on gingival/alveolar mucosa (four cases), hard palate, and floor of the mouth, measuring 17-40 mm (median 31 mm). The duration of the lesions ranged from 3 months to several years. Information on IgG4 serum levels was available for two patients, and these were increased to 1.85 and 1.65 g/L, respectively. The follow-up period ranged 11-30 months (median 13 months). None of the lesions recurred, and none of the patients developed any manifestation of IgG4-RD. Microscopically, all cases presented as nodular lesions composed of numerous polyclonal plasma cells admixed with lymphocytes, histiocytes, mast cells, and eosinophils, set within collagenized stroma in variable proportions. Obliterative phlebitis was observed in two cases. The number of IgG4-positive plasma cells ranged between 51 and 142 per HPF (median 114), while the IgG4/IgG ratio values ranged between 0.16 and 0.72 (median 0.44) and were above 0.40 in three cases. Based on international criteria, two cases were diagnosed as definite and one as probable IgG4-RD. Oral plasma cell granuloma is a heterogeneous group of lesions, and a subset may represent a mucosal manifestation of IgG4-RD in the oral cavity.

摘要

本研究的目的是检验如下假设

口腔浆细胞性肉芽肿可能是口腔中免疫球蛋白(Ig)G4相关疾病(IgG4-RD)的一种黏膜表现。研究样本包括两名男性和四名女性,年龄在54至79岁之间(中位数为62岁)。病变位于牙龈/牙槽黏膜(4例)、硬腭和口底,大小为17至40毫米(中位数为31毫米)。病变持续时间从3个月到数年不等。两名患者有IgG4血清水平信息,其IgG4血清水平分别升高至1.85克/升和1.65克/升。随访期为11至30个月(中位数为13个月)。所有病变均未复发,且所有患者均未出现IgG4-RD的任何表现。显微镜下,所有病例均表现为结节性病变,由大量多克隆浆细胞与淋巴细胞、组织细胞、肥大细胞和嗜酸性粒细胞混合组成,以不同比例分布于胶原化的间质中。两例观察到闭塞性静脉炎。每高倍视野(HPF)IgG4阳性浆细胞数量在51至142之间(中位数为114),而IgG4/IgG比值在0.16至0.72之间(中位数为0.44),三例高于0.40。根据国际标准,两例被诊断为确诊的IgG4-RD,一例为可能的IgG4-RD。口腔浆细胞性肉芽肿是一组异质性病变,其中一部分可能是口腔中IgG4-RD的黏膜表现。

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