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

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 heterogenous group of lesions, and a subset may represent a mucosal manifestation of IgG4-RD in the oral cavity.

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Acknowledgments

The authors thank Mrs. M. Žáková for her excellent technical support, Vratislav Zita, M.D., for providing clinical data in patient no. 5, and Prof. I. Šteiner, M.D., Ph.D. for English language correction. The experiments carried out in this study comply with the current laws of the Czech Republic. This study was supported by the program PRVOUK P37/11 and by the project LM2010004. The funding sources had no involvement on the study design and collection, analysis, and interpretation of data, on the writing of the report, and on the decision to submit the article for publication.

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The authors declare that they have no conflict of interest.

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Correspondence to Jan Laco.

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Laco, J., Kamarádová, K., Mottl, R. et al. Plasma cell granuloma of the oral cavity—a mucosal manifestation of immunoglobulin G4-related disease or a mimic?. Virchows Arch 466, 255–263 (2015). https://doi.org/10.1007/s00428-014-1711-6

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Keywords

  • Oral cavity
  • Plasma cell granuloma
  • Plasma cell gingivitis
  • Inflammatory pseudotumor
  • IgG4-related disease
  • IgG4