Orbital Necrobiotic Xanthogranuloma Associated With Systemic IgG4 Disease

被引:36
作者
Singh, Kamaljeet [2 ]
Rajan, K. D. A. [3 ]
Eberhart, Charles [1 ]
机构
[1] Johns Hopkins Sch Med, Baltimore, MD 21218 USA
[2] Brown Univ, Providence, RI 02912 USA
[3] Johns Hopkins Med Inst, Wilmer Eye Inst, Baltimore, MD 21205 USA
关键词
dacryoadenitis; IGG4; IGG; lymphocytic sclerosing pancreatitis; orbital xanthogranuloma; HEPATIC INFLAMMATORY PSEUDOTUMOR; CHRONIC SCLEROSING SIALADENITIS; RETROPERITONEAL FIBROSIS; DACRYOADENITIS; SPECTRUM; PATIENT; PART;
D O I
10.3109/09273948.2010.490629
中图分类号
R77 [眼科学];
学科分类号
100212 ;
摘要
Purpose: The authors describe 2 cases of orbital xanthogranulomatous disease associated with an increase in IgG4-positive plasma cells, and also examine IgG4 in other types of orbital inflammation. Methods: Immunohistochemistry for total IgG and IgG4 was performed in 18 cases of orbital inflammation, including chronic dacryoadenitis (n=10), necrobiotic xanthogranuloma (n=2), xanthogranuloma (n=1), idiopathic orbital inflammation/pseudotumor (n=4), and fungal infection (n=1). Results: One patient presenting with necrobiotic xanthogranuloma had signs of systemic IgG4 disease. His orbital lesion showed an elevated number of IgG4 positive plasma cells (55%). An orbital xanthogranulomatous lesion in a second patient lacking systemic symptoms also contained a high percentage of IgG4-positive plasma cells (80%). Only 1 case of chronic dacryoadenitis contained prominent IgG4-positive plasma cells (mean 17/hpf). Conclusions: IgG4-positive plasma cells are relatively rare in nonsclerosing orbital inflammatory lesions. However, systemic disease IgG4 can be associated with necrobiotic xanthogranuloma of the orbit.
引用
收藏
页码:373 / 378
页数:6
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