Multiple IgG4‐related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum

Abstract

IgG4-related sclerosing disease is recognized as a distinct clinicopathological entity. It is well known that this disease can occur in the salivary, lacrimal and pituitary glands, in the head and neck region. The nasal cavity is an extremely rare site of involvement of IgG4-related sclerosing disease. Herein is reported a case of multiple IgG4-related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum. A 73-year-old Japanese man presented with nasal obstruction and tumors of the right maxillary sinus and parotid gland were detected, after which resections of these tumors were performed. One year after the last surgery, he noted swelling of the nasal septum, and the tumor was resected. These three tumors had similar histopathology, such as conspicuous fibrosclerotic changes with dense lymphoplasmacytic infiltration and occasional obliterative phlebitis. Immunohistochemistry indicated abundant IgG4-positive plasma cell infiltration and high ratios of IgG4-positive/IgG-positive plasma cells (>70%) in all three lesions. The diagnosis of multiple IgG4-related sclerosing lesions was made. The present case suggests that IgG4-related sclerosing lesion can occur in the maxillary sinus and nasal septum, and represents an extension of the spectrum of IgG4-related sclerosing disease.

IgG4-related disease in the head and neck region: report of two cases and review of the literature

“…Upper respiratory tract presentation of the disease is rare (Table I) [3,8,9,11,12,13,14,15,16]. A review of the available literature showed only single cases of IgG4-RD of the oral cavity with presentation in the palate, tongue, floor of the mouth, and upper and lower gingiva [3,8,9].…”

IgG4-related sclerosing disease clinically mimicking oral squamous cell carcinoma

“…[9][10][11] Recent reports have shown destructive IgG4 lesions involving the maxillary sinus 12 and nasal septum, 13 both of which resolved after treatment with systemic corticosteroids. Gill et al reported salivary duct carcinoma arising in IgG4-related fibrosclerosis involving the parotid gland.…”

Immunoglobulin G4−Related Sclerosing Disease Mimicking Invasive Tumor in the Nasal Cavity and Paranasal Sinuses

“…[7][8][9] In the head and neck region, IgG4RSD commonly manifests as chronic sclerosing sialadenitis, chronic sclerosing dacryoadenitis, and lymphoplasmacytic hypophysitis. 1,10,11 IgG4RSD is considered to result from an imbalance in the regulatory immune reaction, 12 but the mechanisms are still unclear. IgG4RSD develops predominantly in males 13 and involves Ն2 sites in most patients, though some patients may have disease involving only 1 organ.…”

“…There is insufficient radiologic data for IgG4RSD affecting the nasal cavity and paranasal sinuses. [1][2][3] We present a case of IgG4RSD mimicking an invasive malignant tumor in the nasal cavity and maxillary sinuses on both sides.…”

“…1 When it affects the head and neck, it commonly involves the salivary, lachrymal, and pituitary glands. There is insufficient radiologic data for IgG4RSD affecting the nasal cavity and paranasal sinuses.…”