Abstract
IgG4-related disease (IgG4-RD) is characterized by IgG4-positive plasma cell infiltration and fibrosis mostly in the pancreas, bile ducts, salivary glands, and orbits. A 34-year-old woman with a history of chronic rhinitis presented with bilateral asymptomatic orbital swelling of 7 years’ duration. Extra-oral examination revealed bilateral palpebral swelling and dacryoadenitis, whereas bilateral normal-colored swelling of the floor of the mouth was detected intra-orally. Serologic tests for IgG4 showed an elevated concentration of 698 mg/dL, and an incisional oral biopsy was performed. Histopathologic analysis revealed minor salivary glands with marked infiltration of lymphocytes, histiocytes, and plasma cells, with occasional lymphoid follicles. By immunohistochemistry, the histiocytes were positive for CD163, and IgG4 was positive in more than 50 plasma cells per high-power field. The final diagnosis was IgG4-RD. The patient was referred for treatment in a rheumatology service. Systemic involvement was not detected, and there was an improvement of orbital and oral lesions after oral corticosteroid therapy.
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