Immunoglobulin G4-related periodontitis: case report and review of the literature

Abstract

Background Immunoglobulin G4-related disease (IgG4-RD) is a chronic inflammatory systemic disease of unknown etiology that can affect one or multiple organs. The disease can mimic many infectious and inflammatory diseases, mainly causing organ enlargement or hyperplasia. Its diagnosis primarily relies on clinical, serologic, and histological features (lymphoplasmacytic infiltrates, storiform fibrosis, and obliterative phlebitis of IgG4 + plasma cells). Here, we report a rare case of IgG4-related periodontitis, and review the relevant literatures. Case presentation A 38-year-old Chinese man visited the Department of Periodontics with gingival enlargement, loose teeth, and tooth loss. The patient had very poor oral hygiene and a large amount of calculus. Gingivae were edematous with deep periodontal pockets and attachment loss. Panoramic radiography showed alveolar bone loss. Serologic examination showed that IgG was 23.70 g/L and IgG4 concentration was 2.800 g/L. There was significant lymphoplasmacytic infiltration, a storiform pattern of fibrosis, and mitotic figures with hematoxylin and eosin staining; immunohistochemical staining showed 10 scattered IgG4-positive plasma cells in a high-power field. The patient was diagnosed as IgG4-related periodontitis. He received a course of corticosteroids with periodontal therapy, and the enlargement was significantly improved without recurrence. Conclusion IgG4-RD in the oral and maxillofacial region mainly involves salivary glands, but this rare case was characterized by enlarged gingivae. The differential diagnosis of IgG4-RD should be based on the clinical features and serologic (IgG4) and histopathological examinations. Corticosteroid therapy is effective for most IgG4-RD patients. Taken together, we hope this case report and the literature review can help dentists to improve their understanding of the IgG4-RD.

The “great imitator”: IgG4‐related disease of the oral cavity. Two case reports and scoping review

“…38 In two cases, the swelling was more prominent, similar to drug-related gingival hyperplasia or hematological malignancy. 39,40 In one report, lesions appeared as desquamative gingivitis and were associated with buccal mucosa ulcerations, as stated before. 37 Periodontal symptoms, including tooth loss, bleeding on manipulation and pain were common complaints.…”

“…The gums were affected in 5 cases (9.8%), with lesions whose appearance often resembled the signs of PCG, that is, generalized swelling with bright‐red erythema either on both the vestibular and lingual aspects or only on the vestibular one 38 . In two cases, the swelling was more prominent, similar to drug‐related gingival hyperplasia or hematological malignancy 39,40 . In one report, lesions appeared as desquamative gingivitis and were associated with buccal mucosa ulcerations, as stated before 37 .…”

“…The orthopantomogram often revealed horizontal resorption of the alveolar bone in gingival cases, similar to the picture observed in periodontal disease. 39 CT scans of lesions involving the jaw bones only reinforced the suspicion of malignancy or infection, rather than identifying features that could make clinicians lean to a diagnosis of IgG4-RD. 27 MRI helped to better visualize the pseudotumors in soft tissues, but a nonhomogeneous signal on T2-weighted slides and an irregular enhancement after cmi were commonly reported, corroborating thus the suspicion of a neoplastic disease.…”

Oral and maxillofacial manifestations of IgG4‐related disease: A clinicopathological study

“…In general, the natural history of IgG4‐RD is unpredictable, ranging from spontaneous resolution to a high risk of death. Steroids are the standard treatment, usually leading to an improvement of the condition, 14,16 although relapse is a common event 11 . Alternatives to steroid monotherapy have been reported, including its combination with immunosuppressive drugs such as azathioprine, methotrexate, cyclophosphamide, and mycophenolate mofetil 11 .…”

Gingival plasma cell granuloma: The importance of histological analysis

“…18,19 There have also been clinical similarities noted between PCGs and localised gingival enlargement seen in immunoglobulin G4-related disease (IgG4-RD). 20 Plasma cell granulomas can also be accompanied by a high serum concentration of IgG4 and IgG4-positive plasma cells, but histological analysis would reveal no lymphoplasmacytic infiltrate, storiform fibrosis, or obliterative phlebitis in PCG, as seen in IgG4-RDs. 21 The finding of IgG4-positive plasma cells therefore must be interpreted in context of the histological analysis to avoid misdiagnosis of IgG4-RD.…”