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Immunoglobulin G4 sinusitis in association with aspirin-exacerbated respiratory disease

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Background:

Immunoglobulin G4 (IgG4) related disease is a systemic inflammatory disease characterized by tumor-like tissue infiltration with IgG4 positive (IgG4+) plasma cells. Aspirin-exacerbated respiratory disease (AERD) is defined as asthma, chronic rhinosinusitis with nasal polyposis, and hypersensitivity to cyclooxygenase-1 inhibitors.

Objective:

We described a case of a non-smoking 61-year-old male with prior NSAID sensitivity who presented with a 1-year history of left eye proptosis associated with chronic nasal symptoms, ultimately identified as concurrent AERD and IgG4 sinusitis.

Methods:

The patient was evaluated in the clinic and diagnosed by using clinical, radiographic, and surgical biopsy findings.

Results:

Although initial concern was greatest for malignancy, a biopsy specimen confirmed the presence of a dense lymphoplasmacytic infiltrate and storiform fibrosis, associated with increased IgG4+ plasma cells. Therefore, IgG4‐related disease (RD) was identified in this patient with AERD.

Conclusion:

Shared type II inflammation may be responsible for the coexistence of IgG4-RD and AERD as observed in our patient. Health care workers must be cognizant of the simultaneous presentation of both IgG4-RD and AERD.
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Keywords: Anosmia; IgG4-related disease; NSAID sensitivity; aspirin exacerbated respiratory disease; eosinophilia; lymphoplasmacytic infiltration; proptosis; sinusitis; type 2 inflammation

Document Type: Research Article

Publication date: 2017-09-01

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