IgG4-related disease presenting with inflammatory pseudotumor of the buccal submucosa: A case report. Issue 2 (March 2018)

• Record Type: Journal Article
• Authors: Kobashi, H.; Ishii, S.; Yakushiji, N.
• Is Part Of: Journal of oral and maxillofacial surgery, medicine, and pathology. Volume 30:Issue 2(2018)
• Page Start: 166
• Page End: 168
• Publication Date: 2018-03
• Subjects: Inflammatory pseudotumor (IPT) -- Immunoglobulin G4-related disease (IgG4-RD) -- IgG4-positive plasma cells; Mouth -- Surgery -- Periodicals; Face -- Surgery -- Periodicals; Maxilla -- Surgery -- Periodicals; Oral medicine -- Periodicals; Mouth -- Diseases -- Pathogenesis -- Periodicals; Surgery, Oral -- Periodicals; Oral Medicine -- Periodicals; Pathology, Oral -- Periodicals; Face -- Surgery; Maxilla -- Surgery; Mouth -- Diseases -- Pathogenesis; Mouth -- Surgery; Oral medicine; Electronic journals -- Sciences; Electronic journals -- Medicine; Periodicals; 617.522059
• DOI: 10.1016/j.ajoms.2017.11.005
• Languages: English
• ISSNs: 2212-5566

Abstract

Abstract: Inflammatory pseudotumor is a rare clinical condition that may be associated with immunoglobulin G4-related disease. However, conclusive etiological links with immunoglobulin G4-related disease have not been identified in the few reported cases of oral inflammatory pseudotumors. We describe a case of an inflammatory pseudotumor of the buccal submucosa that was histopathologically confirmed to be a manifestation of immunoglobulin G4-related disease. The patient was an 83-year-old man who visited our department for the examination of swelling in the left buccal mucosa. The initial examination identified a painless, movable tumor with a long axis (approximately 40 mm) on the left buccal submucosa. Biopsy, magnetic resonance imaging, and computed tomographic findings did not clearly indicate malignancy, and the tumor was excised to facilitate a confirmative diagnosis. Histopathological examination revealed inflammatory cell infiltration of the adipose tissue as well as many lymphoid follicles. Immunohistochemistry predominantly revealed immunoglobulin G4-related disease-positive cells, thereby confirming the diagnosis of immunoglobulin G4-related disease. However, a postoperative blood test revealed normal immunoglobulin G4 levels, and steroid therapy was not initiated. The patient remains symptom- and recurrence-free at 18 months after surgery.