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

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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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References

  1. Ide F, Shimoyama T, Kato T, Horie N, Kusama K (2000) Inflammatory pseudotumor and related lesions of the oral and maxillofacial region: a critical reappraisal based on recent information. Oral Med Pathol 5:41–47

    Article  Google Scholar 

  2. Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F (2013) WHO classification of tumours of soft tissue and bone. IARC, Lyon, pp 83–84

    Google Scholar 

  3. Rosai J (2011) Rosai and Ackerman’s surgical pathology. Mosby, p. 251

  4. Ballesteros E, Osborne BM, Matsushima AY (1998) Plasma cell granuloma of the oral cavity: a report of two cases and review of the literature. Mod Pathol 11:60–64

    CAS  PubMed  Google Scholar 

  5. Pandav AB, Gosavi AV, Lanjewar DN, Jagadale RV (2012) Gingival plasma cell granuloma. Dent Res J 9:816–820

    Google Scholar 

  6. Manohar B, Bhuvaneshwari S (2011) Plasma cell granuloma of gingiva. J Indian Soc Periodontol 15:64–66. doi:10.4103/0972-124X.82275

    Article  PubMed Central  PubMed  Google Scholar 

  7. Phadnaik MB, Attar N (2010) Gingival plasma cell granuloma. Indian J Dent Res 21:460–462. doi:10.4103/0970-9290.70788

    Article  PubMed  Google Scholar 

  8. Laco J (2010) IgG4-related systemic sclerosing disease: a review. Cesk Patol 46:82–85

    CAS  PubMed  Google Scholar 

  9. Hrncir Z, Laco J (2011) Clinical importance of the IgG4-related disease. Cas Lek Cesk 150:438–441

    PubMed  Google Scholar 

  10. Stone JH, Zen Y, Deshpande V (2012) IgG4-related disease. N Engl J Med 366:539–551. doi:10.1056/NEJMra1104650

    Article  CAS  PubMed  Google Scholar 

  11. Ferry JA, Deshpande V (2012) IgG4-related disease in the head and neck. Sem Diagn Pathol 29:235–244. doi:10.1053/j.semdp.2012.07.008

    Article  Google Scholar 

  12. Bhatti RM, Stelow EB (2013) IgG4-related disease of the head and neck. Adv Anat Pathol 20:10–16. doi:10.1097/PAP.0b013e31827b619e

    Article  CAS  PubMed  Google Scholar 

  13. Agaimy A, Ihrler S (2014) Immunoglobulin G4 (IgG4)-related disease. A review of head and neck manifestations. Pathologe 35:152–159. doi:10.1007/s00292-013-1848-0

    Article  CAS  PubMed  Google Scholar 

  14. Laco J, Ryska A, Celakovsky P, Dolezalova H, Mottl R, Tucek L (2011) Chronic sclerosing sialadenitis as one of the immunoglobulin G4-related diseases: a clinicopathological study of six cases from Central Europe. Histopathology 58:1157–1163. doi:10.1111/j.1365-2559.2011.03833.x

    Article  PubMed  Google Scholar 

  15. Hrncir Z, Laco J, Slezak R, Rencova E, Drahosova M, Brtkova J (2011) Mikulicz’s disease with unilateral exophthalmia—IgG4-related disease. Ces Revmatol 19:125–130

    Google Scholar 

  16. Cameselle-Teijeiro J, Ladra MJ, Abdulkader I, Eloy C, Soares P, Barreiro F et al (2014) Increased lymphangiogenesis in riedel thyroiditis (immunoglobulin G4-related thyroid disease). Virchows Arch 465:359–364. doi:10.1007/s00428-014-1626-2

    Article  CAS  PubMed  Google Scholar 

  17. Li Y, Zhou G, Ozaki T, Nishihara E, Matsuzuka F, Bai Y et al (2012) Distinct histopathological features of Hashimoto’s thyroiditis with respect to IgG4-related disease. Mod Pathol 25:1086–1097. doi:10.1038/modpathol.2012.68

    Article  PubMed  Google Scholar 

  18. Deshpande V, Khosroshahi A, Nielsen GP, Hamilos DL, Stone JH (2011) Eosinophilic angiocentric fibrosis is a form of IgG4-related systemic disease. Am J Surg Pathol 35:701–706. doi:10.1097/PAS.0b013e318213889e

    Article  PubMed  Google Scholar 

  19. Cheuk W, Tam FKY, Chan ANH, Luk ISC, Yuen APW, Chan WK et al (2010) Idiopathic cervical fibrosis—a new member of IgG4-related sclerosing disease: report of 4 cases, 1 complicated by composite lymphoma. Am J Surg Pathol 34:1678–1685. doi:10.1097/PAS.0b013e3181f12c85

    PubMed  Google Scholar 

  20. Cheuk W, Chan JKC (2012) Lymphadenopathy of IgG4-related disease: an underdiagnosed and overdiagnosed entity. Semin Diagn Pathol 29:226–234. doi:10.1053/j.semdp.2012.07.001

    Article  PubMed  Google Scholar 

  21. Ishida M, Hotta M, Kushima R, Shibayama M, Shimizu T, Okabe H (2009) Multiple IgG4-related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum. Pathol Int 59:670–675. doi:10.1111/j.1440-1827.2009.02425.x

    Article  PubMed  Google Scholar 

  22. Ikeda R, Awataguchi T, Shoji F, Oshima T (2010) A case of paranasal sinus lesions in IgG4-related sclerosing disease. Otolaryngol Head Neck Surg 142:458–459. doi:10.1016/j.otohns.2009.09.019

    Article  PubMed  Google Scholar 

  23. Pace C, Ward S (2010) A rare case of IgG4-related sclerosing disease of the maxillary sinus associated with bone destruction. J Oral Maxillofac Surg 68:2591–2593. doi:10.1016/j.joms.2009.07.073

    Article  PubMed  Google Scholar 

  24. Cho HK, Lee YJ, Chung JHC, Koo JW (2011) Otologic manifestation in IgG4-related systemic disease. Clin Exp Otorhinolaryngol 4:52–54. doi:10.3342/ceo.2011.4.1.52

    Article  PubMed Central  PubMed  Google Scholar 

  25. Khurram SA, Fernando M, Smith AT, Hunter KD (2013) IgG4-related sclerosing disease clinically mimicking oral squamous cell carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol 115:e48–e51. doi:10.1016/j.oooo.2012.04.011

    Article  PubMed  Google Scholar 

  26. Lindau RH, Su YB, Kobayashi R, Smith RB (2013) Immunoglobulin G4-related sclerosing disease of the paranasal sinus. Head Neck 35:E321–E324. doi:10.1002/hed.23175

    Article  PubMed  Google Scholar 

  27. Andrew N, Kearney D, Sladden N, Goss A, Selva D (2014) Immunoglobin G4-related disease of the hard palate. J Oral Maxillofac Surg 72:717–723. doi:10.1016/j.joms.2013.08.033

    Article  PubMed  Google Scholar 

  28. Khoo JF, Batt M, Stimpson P, Safdar A (2014) Supraglottic immunoglobulin-G4 related plasma cell granuloma: case report and literature review. Head Neck 36:E57–E59. doi:10.1002/hed.23519

    Article  PubMed  Google Scholar 

  29. Laco J, Steiner I, Holubec T, Dominik J, Holubcova Z, Vojacek J (2011) Isolated thoracic aortitis: clinicopathological and immunohistochemical study of 11 cases. Cardiovasc Pathol 20:352–360. doi:10.1016/j.carpath.2010.09.003

    Article  CAS  PubMed  Google Scholar 

  30. Laco J, Podhola M, Kamarádová K, Novák I, Dobeš D, Broďák M et al (2013) Idiopathic vs. secondary retroperitoneal fibrosis: a clinicopathological study of 12 cases, with emphasis to possible relationship to IgG4-related disease. Virchows Arch 463:721–730. doi:10.1007/s00428-013-1480-7

    Article  CAS  PubMed  Google Scholar 

  31. Umehara H, Okazaki K, Masaki Y et al (2012) Comprehensive diagnostic criteria for IgG4-related disease (IgG4-RD), 2011. Mod Rheumatol 22:21–30. doi:10.1007/s10165-011-0571-z

    Article  CAS  PubMed  Google Scholar 

  32. Laco J, Örhalmi J, Bártová J, Zimandlová D (2014) Enterocolic lymphocytic phlebitis as a newly recognized manifestation of IgG4-related disease. Int J Surg Pathol (in press)

  33. Deshpande V, Zen Y, Chan JKC et al. (2012) Consensus statement on the pathology of IgG4-related disease. Mod Pathol 25:1181–1192. doi: 10.1038/modpathol.2012.72

  34. Strehl JD, Hartmann A, Agaimy A (2011) Numerous IgG4-positive plasma cells are ubiquitous in diverse localized non-specific chronic inflammatory conditions and need to be distinguished from IgG4-related disorders. J Clin Pathol 64:237–243. doi:10.1136/jcp.2010.085613

    Article  PubMed  Google Scholar 

  35. Šteiner I, Krbal L, Rozkoš T, Harrer J, Laco J (2012) Calcific aortic valve stenosis: immunohistochemical analysis of inflammatory infiltrate. Pathol Res Pract 208:231–234. doi:10.1016/j.prp.2012.02.009

    Article  PubMed  Google Scholar 

  36. Tošovský M, Bradna P, Laco J, Podhola M, Soukup T, Brožík J (2012) ANCA associated glomerulonephritis in combination with IgG4-positive mediastinal mass in a patient with ankylosing spondylitis treated with TNF alpha inhibitors. Acta Med (Hradec Kralove) 55:42–46

    Google Scholar 

  37. Chang SY, Keogh KA, Lewis JE, Ryu JH, Cornell LD, Garrity JA et al (2013) IgG4-positive plasma cells in granulomatosis with polyangiitis (Wegener’s): a clinicopathologic and immunohistochemical study on 43 granulomatosis with polyangiitis and 20 control cases. Hum Pathol 44:2432–2437. doi:10.1016/j.humpath.2013.05.023

    Article  CAS  PubMed  Google Scholar 

  38. Tian W, Yakirevich E, Matoso A, Gnepp DR (2012) IgG4(+) plasma cells in sclerosing variant of mucoepidermoid carcinoma. Am J Surg Pathol 36:973–979. doi:10.1097/PAS.0b013e318258f018

    Article  PubMed  Google Scholar 

  39. Lafzi A, Farahani RM, Shoja MA (2006) Amlodipine-induced gingival hyperplasia. Med Oral Patol Oral Cir Bucal 11:E480–E482

    PubMed  Google Scholar 

  40. Vishnudas B, Sameer Z, Shriram B, Rekha K (2014) Amlodipine induced plasma cell granuloma of the gingiva: a novel case report. J Nat Sci Biol Med 5:472–476. doi:10.4103/0976-9668.136267

    Article  PubMed Central  PubMed  Google Scholar 

  41. Yamamoto H, Yamaguchi H, Aishima S, Oda Y, Kohashi K, Oshiro Y et al (2009) Inflammatory myofibroblastic tumor versus IgG4-related sclerosing disease and inflammatory pseudotumor: a comparative clinicopathologic study. Am J Surg Pathol 33:1330–1340

    Article  PubMed  Google Scholar 

  42. Saab ST, Hornick JL, Fletcher CD, Olson SJ, Coffin CM (2011) IgG4 plasma cells in inflammatory myofibroblastic tumor: inflammatory marker or pathogenic link? Mod Pathol 24:606–612. doi:10.1038/modpathol.2010.226

    Article  CAS  PubMed  Google Scholar 

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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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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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  • DOI: https://doi.org/10.1007/s00428-014-1711-6

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