IgG4-related disease presenting with inflammatory pseudotumor of the buccal submucosa: A case report

Kobashi, Haruhiko; Ishii, Shiro; Yakushiji, Noboru

doi:10.1016/j.ajoms.2017.11.005

Cited by 1 publication

(4 citation statements)

References 12 publications

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“…As lesions on the buccal mucosa, they showed a homogeneous appearance, usually with submucosal swellings, similar to IPT or minor salivary gland neoplasms 35,36 . Only one paper reported the case of a mucosal ulceration resembling erosive oral lichen planus, but it was also associated with gingival findings 37 …”

Section: Scoping Review: Resultsmentioning

confidence: 99%

“…21,56,57 For lesions in the buccal mucosa, on the other hand, soft tissue and minor salivary gland tumors must be ruled out. 35,36 A further detailed discussion would be necessary about those lesions affecting gingival tissues, where they usually appear identical to PCG or may resemble drugrelated gingival hyperplasia. 38,39 IgG4-RD is renowned for multiple organ involvement: most oral cases are not isolated in the mouth, but occur with synchronous or metachronous extraoral lesions, especially in the head and neck, with the salivary glands, lacrimal glands, sinonasal tract, and cervical lymph nodes being the preferred sites.…”

Section: Discussionmentioning

confidence: 99%

“…In these locations differential diagnosis is made with the more aggressive inflammatory myofibroblastic tumor and, for those cases with masses radiating from the maxilla to the paranasal sinuses, it is necessary to exclude actinomycosis, fungal infection, or malignancy 21,56,57 . For lesions in the buccal mucosa, on the other hand, soft tissue and minor salivary gland tumors must be ruled out 35,36 …”

Section: Discussionmentioning

confidence: 99%

“…As lesions on the buccal mucosa, they showed a homogeneous appearance, usually with submucosal swellings, similar to IPT or minor salivary gland neoplasms. 35,36 Only one paper reported the case of a mucosal ulceration resembling erosive oral lichen planus, but it was also associated with gingival findings. 37 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.…”

Section: Clinical Presentationmentioning

confidence: 99%

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

Azzi,

Magnoli,

Krepysheva

et al. 2024

Head & Neck

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This study aimed to review the lesser‐known intraoral manifestations of immunoglobulin G4‐related disease (IgG4‐RD). In this paper we report an unprecedented case of oral IgG4‐RD mimicking angiolymphoid hyperplasia with eosinophilia (ALHE), and another case presenting as plasma cell gingivitis. We then performed a scoping review of published cases of IgG4‐RD involving the oral cavity. The following data were collected for each case: age, sex, intraoral site(s) involved, clinical appearance, imaging features, serum IgG4 values, histopathology, treatment, and follow‐up duration. Fifty‐one cases of oral IgG4‐RD were published in literature. The hard palate and jaw bones were the two main locations reported, while the histological identification of a IgG4/IgG plasma cells ratio ≥40% was fundamental for diagnosis. Conversely, the pathological features of storiform fibrosis and obliterative phlebitis were not common. Future reports regarding oral IgG4‐RD should report clear adherence to the recognized international diagnostic criteria of the disease.

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Section: Scoping Review: Resultsmentioning

confidence: 99%