Minor salivary IgG-4 related sialadenitis – Clinical and diagnostic challenges

Cohen, Oded; Azoulay, Ofer; Feldberg, Edit; Sthoeger, Zev; Schindel, Doron; Halperin, Doron

doi:10.1016/j.ajoms.2017.04.002

Cited by 5 publications

(5 citation statements)

References 12 publications

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“…Precisely, the hard palate was the site most frequently affected by oral IgG4‐RD, accounting for 29.4% of individuals (15/51). In this region, lesions presented as bilateral, red‐purple, rubbery, and boggy swellings associated with discomfort due to local enlargement (9/15), while in two cases lesions appeared as submucosal nodules with more defined boundaries 25,26 . In a single report, palatal IgG4‐RD showed midline destructive lesions (MDL), with perforation and involvement of nasal structures, similar to what observed in GPA or cocaine‐induced MDL (4/15) 24 .…”

Section: Scoping Review: Resultsmentioning

confidence: 74%

“…In this region, lesions presented as bilateral, red-purple, rubbery, and boggy swellings associated with discomfort due to local enlargement (9/15), while in two cases lesions appeared as submucosal nodules with more defined boundaries. 25,26 In a single report, palatal IgG4-RD showed midline destructive lesions (MDL), with perforation and involvement of nasal structures, similar to what observed in GPA or cocaine-induced MDL (4/15). 24 However, it was highlighted that these destructive lesions had been progressively worsening over 2-3 years, thus they represented long-standing lesions that had evolved in MDL.…”

Section: Clinical Presentationmentioning

confidence: 71%

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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: 74%

Section: Clinical Presentationmentioning

confidence: 71%

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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“…Microscopic findings have a great significance in the diagnosis of this disease, given that radiological imaging is unable to distinguish IgG4-RD from other conditions such as malignant tumours and other similar disorders. 21,23 The histological diagnosis depends on the identification of the triad: dense lymphoplasmacytic infiltrate, with germinal centres occasionally observed, storiform fibrosis, and obliterative phlebitis, which is the least commonly identified histologic feature. 6 Other minor histopathological findings to be considered are the mildto-moderate tissue eosinophilia and phlebitis without obliteration of the lumen.…”

Section: Discussionmentioning

confidence: 99%

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

Pereira

Pontes

Soares

et al. 2022

J Oral Pathology Medicine

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Background IgG4‐related disease is a fibroinflammatory and immune‐mediated condition, which has extremely variable clinical manifestations. In this study, we aim to investigate the clinicopathological features of IgG4‐related disease involving the oral and maxillofacial region. Methods Cases of IgG4‐related disease manifesting in the oral and maxillofacial region were retrieved from three Brazilian institutions. Clinical and serological data were obtained from the patients' medical charts, while microscopic and immunohistochemical findings were revised by oral pathologists. Diagnosis followed the American College of Rheumatology/European League against Rheumatism criteria. Results Seven patients diagnosed with IgG4‐related disease were included in this study. Women were affected in all analysed cases, with a mean age of 55.4 years. Two patients presented with the clinical involvement of more than one oral and maxillofacial anatomic site. Therefore, our sample comprised nine oral and maxillofacial anatomic sites affected by IgG4‐related disease. The submandibular gland was affected in four cases, the tongue and the parotid gland in two cases each, and the palate in one case. In a few cases, exploratory lower lip biopsy was used as a diagnostic approach. A moderate‐to‐severe lymphoid infiltrate containing plasma cells and lymphocytes, with an increased IgG4/IgG ratio, was common. Treatment varied and steroids were the most frequently used (57.4%). Six patients remained alive, while one died from unknown causes. Conclusion Although major salivary glands are commonly affected by IgG4‐related disease, the oral cavity can also be involved, and lower lip biopsy may be an auxiliary diagnostic tool.

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“…FNAC was a limited tool to obtain a valid and conclusive sample to evaluate IgG4 positive plasma cells. Therefore, it is very important that the surgeon can obtain an adequate biopsy sample with enough tissue to carry out a correct pathological evaluation (12). However, the incisional biopsy performed was unable to initially diagnose the presence of an occult tumor foci that was hidden within IgG4-RS involvement.…”

Section: Discussionmentioning

confidence: 99%

Synchronous occurrence of IgG4-related sialadenitis and ductal carcinoma of the parotid gland: a case report

Mayordomo¹,

Camarena²,

Gutiérrez-Domingo³

et al. 2021

Gland Surg

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Immunoglobulin G4-related disease (IgG4-RD) is a rare chronic systemic inflammatory pathology that poses a diagnostic challenge since it can simulate malignancy when it affects a salivary gland as a mass-like lesion. Here, the authors report an unusual clinical case of a 42-year-old man who presented with a painless, slow-growing swelling located in the right parotid gland with a 12-month evolution. Based on imaging tests and open biopsy, a diagnosis of chronic parotitis was presumed and oral methylprednisolone was prescribed. Due to poor response to medication, a total parotidectomy preserving the facial nerve was performed. The final pathology described a unilateral IgG4-related sialadenitis (IgG4-RS) in the parotid gland in combination with a poorly differentiated multifocal ductal carcinoma. The postoperative course was uneventful except for a temporary facial paresis (grade III according to the House-Brackmann classification system) that resolved completely within 5 months. There were no systemic manifestations on the whole-body 18F-FDG PET/CT. Adjuvant radiotherapy was administered without complications. Twenty-four months follow-up after surgery showed no recurrence or evidence of systemic involvement. This clinical report highlights the importance of considering the synchronous occurrence of a carcinoma underlying an isolated parotid gland mass in the context of IgG4-RS, especially if there is no response to prior steroid medication.

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Minor salivary IgG-4 related sialadenitis – Clinical and diagnostic challenges

Cited by 5 publications

References 12 publications

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

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

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

Synchronous occurrence of IgG4-related sialadenitis and ductal carcinoma of the parotid gland: a case report

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