Common Characteristics of Sinonasal Inflammation Associated with IgG4-Related Disease and Other Chronic Inflammatory Diseases: A Retrospective Observational Study

Ryosuke Sato; Takumi Kumai; Ryota Yuasa; Risa Wakisaka; Hiroki Komatsuda; Hidekiyo Yamaki; Yoshiya Ishida; Tetsuji Wada; Miki Takahara

doi:10.1159/000538365

Common Characteristics of Sinonasal Inflammation Associated with IgG4-Related Disease and Other Chronic Inflammatory Diseases: A Retrospective Observational Study

ORL J Otorhinolaryngol Relat Spec. 2024 Apr 25:1-6. doi: 10.1159/000538365. Online ahead of print.

Authors

Ryosuke Sato^{1

2}, Takumi Kumai^{2

3}, Ryota Yuasa², Risa Wakisaka², Hiroki Komatsuda², Hidekiyo Yamaki², Yoshiya Ishida¹, Tetsuji Wada¹, Miki Takahara^{2

3}

Affiliations

¹ Department of Otolaryngology, Head and Neck Surgery, Kitami Red Cross Hospital, Kitami, Japan.
² Department of Otolaryngology-Head and Neck Surgery, Asahikawa Medical University, Asahikawa, Japan.
³ Department of Innovative Head and Neck Cancer Research and Treatment, Asahikawa Medical University, Asahikawa, Japan.

PMID: 38663371
DOI: 10.1159/000538365

Abstract

Introduction: Immunoglobulin G4-related disease (IgG4-RD) is a systemic inflammatory disease characterized by elevated serum IgG4, tissue infiltration of IgG4-positive cells, and fibrosis. Although a number of IgG4-RD patients show sinonasal involvement, there is little known about sinonasal inflammation associated with IgG4-RD. This study aimed to describe the clinicopathological features of sinonasal inflammation associated with IgG4-RD and to compare with other inflammatory diseases, such as eosinophilic chronic rhinosinusitis (ECRS) and granulomatosis with polyangiitis (GPA).

Methods: A retrospective analysis of clinicopathological features of patients with sinonasal lesions and high serum IgG4 was performed. Patient data were reviewed to determine whether they fulfilled the diagnostic criteria for other inflammatory diseases.

Results: Six of 7 patients were diagnosed with IgG4-RD, while 1 patient was diagnosed with GPA. In the 6 patients with IgG4-RD, intranasal findings showed nasal polyps in 3 patients (50%) and nasal crusting in the 3 patients (50%). Computed tomography showed ethmoid sinus involvement in 5 patients (83%). Five of the 6 patients (83%) were diagnosed with IgG4-RD based on nasal biopsy, whereas 1 patient (17%) was diagnosed based on lacrimal gland biopsy. Four patients fulfilled the Japanese epidemiological survey of refractory ECRS (JESREC) criteria. However, none of the patients showed eosinophil infiltration. Although the patient with GPA showed high levels of serum IgG4 and tissue infiltration of IgG4-positive cells in the nasal biopsy, the patient showed common clinical features of GPA.

Conclusion: Patients with sinonasal inflammation associated with IgG4-RD had similar clinical characteristics with ECRS and GPA. Histopathological findings of the nasal biopsy from clinically diagnosed GPA was consistent with that of IgG4-RD. Sinonasal inflammation associated with IgG4-RD should be diagnosed based not only on tissue infiltration of IgG4-positive cells but in conjunction with clinical findings such as local nasal characteristics, involvement of other organs, and serum antineutrophil cytoplasmic antibody levels. IgG4-RD should be ruled out in patients with eosinophilia without histopathological eosinophil infiltration.

Keywords: Antineutrophil cytoplasmic antibody-associated vasculitis; Eosinophilic chronic rhinosinusitis; Granulomatosis with polyangiitis; IgG4-related disease; Sinonasal lesions.