| IgG4-related Ophthalmic Disease Associated with Adult Xanthogranulomatous Disease. |
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Seunghyun Lee, Sokjoong Chung, Jinhyung Heo, Helen Lew |
1Department of Ophthalmology, CHA Bundang Medical Center, Seongnam, Korea. eye@cha.ac.kr
2Department of Pathology, CHA Bundang Medical Center, Seongnam, Korea. |
| 황색육아종과 동반된 면역글로불린G4관련안질환 1예 |
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이승현1⋅정석중1⋅허진형2⋅유혜린1 |
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차의과학대학교 분당차병원 안과학교실1, 차의과학대학교 분당차병원 병리학교실2 |
Correspondence:
Helen Lew,
Email: eye@cha.ac.kr
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Received: 12 July 2018 • Revised: 20 August 2018 • Accepted: 21 October 2018 |
| Abstract |
PURPOSE
To report a case of immunoglobulin G4 (IgG4)-related ophthalmic disease associated with adult xanthogranulomatous disease. CASE SUMMARY: A 38-year-old male with a history of cholecystectomy visited our clinic for bilateral periorbital swelling. Histopathology of the orbital biopsy showed diffuse infiltration of foamy histiocytes with Touton giant cells and lymphoid follicles, with a diagnosis of adult-onset xanthogranuloma. After excisional biopsy, he was treated with azathioprine and prednisolone. Four years after treatment, he again visited the clinic due to bilateral, yellowish eyelid masses. Serological examinations were all nonspecific findings, except for elevation of IgG and IgG4 levels. Magnetic resonance imaging showed bilateral symmetric soft tissue enlargement with slightly heterogeneous T1/T2 isosignal intensity, with contrast enhancement at the superolateral aspect of extraconal spaces. Excisional biopsy and blepharoplasty were performed. Immunohistochemical sections showed that the IgG4+/IgG plasma cell ratio was 10–20% and the IgG4 plasma cell count was 22/high power field (HPF). His past sections of 2013 from the pathology department were again stained and showed that the IgG4+/IgG plasma cell ratio was 40–50% and the IgG4 plasma cell count was 59/HPF. Thus, he was definitely diagnosed with IgG4-related ophthalmic disease. CONCLUSIONS: If there is recurrent eyelid swelling, IgG4-related ophthalmic disease should be considered as a differential diagnosis. And the patient with adult xanthogranulomatous disease can be diagnosed with IgG4-related ophthalmic disease. |
| Key Words:
Adult xanthogranulomatous disease;IgG4-related ophthalmic disease;Xanthogranuloma |
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