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.

The "great imitator": IgG4-related disease of the oral cavity. Two case reports and scoping review

Azzi, Lorenzo
Primo
;
Magnoli, Francesca;Krepysheva, Diana;Fontana, Federico;Coppola, Andrea;Dani, Marta;Battaglia, Paolo;Rabbiosi, Dimitri
2024-01-01

Abstract

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.
2024
IgG4‐RD; IgG4‐related disease; angiolymphoid hyperplasia with eosinophilia; oral IgG4‐RD; plasma cell gingivitis
Azzi, Lorenzo; Magnoli, Francesca; Krepysheva, Diana; Fontana, Federico; Coppola, Andrea; Cappelli, Antonella; Dani, Marta; Battaglia, Paolo; Rabbiosi, Dimitri
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11383/2170191
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