비출혈을 주소로 내원한 환자에서 확인된 비부비동 IgG4-연관 질환 1예
A Case of IgG4 Related Disease in the Nasal Cavity and Paranasal Sinus : A Rare Cause of Nasal Bleeding
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Immunoglobulin (Ig) G4-related disease is a systemic chronic inflammatory disease characterized by sclerosing lesions by IgG4 positive plasma cells and T lymphocytes. It is rarely described in the sinonasal cavity and the paranasal sinus. Here we report a case involving a 69-year-old woman whose chief complaint was recurrent nasal bleeding for 2 years. Her nasal cavity showed erosion of nasal septum as well as the synechia in between the nasal septum and the lateral nasal wall with severe edematous change in the left inferior turbinate. Imaging finding showed bilateral soft tissue density in the maxillary and ethmoid sinuses and mucosal swelling of left nasal cavity. Her IgG4 level was elevated and proteinase-3 Ab was positive. Histopathological examination from the left inferior turbinate showed dense lymphoplasma cell infiltration and storiform fibrosis. Immunostaining for IgG4 and IgG revealed numerous plasma cells positive for IgG4 (more than 90 cells per high power field) and IgG4/IgG ratio of 0.7. Otorhinolaryngologist should consider IgG4 related disease as one of the differential diagnosis in case of bloody rhinorrhea with mucosal erosion or synechia inside the nasal cavity.
Abstract
Immunoglobulin (Ig) G4-related disease is a systemic chronic inflammatory disease characterized by sclerosing lesions by IgG4 positive plasma cells and T lymphocytes. It is rarely described in the sinonasal cavity and the paranasal sinus. Here we report a case involving a 69-year-old woman whose chief complaint was recurrent nasal bleeding for 2 years. Her nasal cavity showed erosion of nasal septum as well as the synechia in between the nasal septum and the lateral nasal wall with severe edematous change in the left inferior turbinate. Imaging finding showed bilateral soft tissue density in the maxillary and ethmoid sinuses and mucosal swelling of left nasal cavity. Her IgG4 level was elevated and proteinase-3 Ab was positive. Histopathological examination from the left inferior turbinate showed dense lymphoplasma cell infiltration and storiform fibrosis. Immunostaining for IgG4 and IgG revealed numerous plasma cells positive for IgG4 (more than 90 cells per high power field) and IgG4/IgG ratio of 0.7. Otorhinolaryngologist should consider IgG4 related disease as one of the differential diagnosis in case of bloody rhinorrhea with mucosal erosion or synechia inside the nasal cavity.
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