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題 名 | Rosai-Dorfman Disease Manifesting as Relapsing Uveitis and Subconjunctival Masses=Rosai-dorfman病--以復發性的葡萄膜炎及結膜下腫瘤表現 |
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作 者 | 譚欣媛; 高玲玉; | 書刊名 | 長庚醫學 |
卷 期 | 25:9 2002.09[民91.09] |
頁 次 | 頁621-625 |
分類號 | 416.74 |
關鍵詞 | Rosai-dorfman病; 葡萄膜炎; 結膜下腫瘤; Rosai-dorfman disease; Uveitis; Subconjunctival mass; |
語 文 | 英文(English) |
中文摘要 | Rosai-Dorfman病是種罕見的組織球細胞增生疾病,主要好發於年輕族群。其主要臨床表徵為出現雙側性的頸部淋巴腫。出現淋巴結外病灶的機率約為28-43%。而出現眼部表徵的機會較為稀少,通常是以侵犯眼窩組織及眼瞼為主。而以葡萄膜炎為表現又更為稀少。我們提出一位63歲Rosai-Dorman病的個案, 以罕見的復發性葡萄膜炎以及結膜下腫瘤作為其臨床表?。病理結果符合Rosai-Dorman病的特徵:出現S100陽性反應的組織球聚集以及淋巴球聚嗜現象。病患臨床病程是復發性但屬良性病程,對於類固醇治療有不錯的反應。我們提出這罕見的病例藉以提醒這全身性的疾病-Rosai-Dorman病可能會以復發性的葡萄膜炎作為其臨床表徵。 |
英文摘要 | Rosai-Dorfman disease (sinus histiocytosis with massive lymphadenopathy, SHML) is a rare, non-hereditary, benign histiocytic proliferative disorder mainly affecting young people with common clinical characteristics such as painless bilateral cervical lymphadenopathy, fever, leukocytosis and polyclonal gammopathy. Extranodal manifestations have been reported in 28-43% of cases. Eye involvement is relatively uncommon (8.5%), and most of cases have presented as lymphoproliferation in the soft tissues of the orbit and eyelids. Uveitis is an even more rare presentation as a review of all the literature. We describe a 63-year-old man with SHML with unusual ophthalmic manifestations of relapsing uveitis and bilateral subconjunctival masses. The results of biopsies were compatible with the characteristic histopatholgical findings of SHML: focal aggregations of S100-positive foamy histiocytes and the existence of lymphocytophagocytosis. During the clinical course, the patient relapsed but the relapse was relatively benign and the patient showed fair response to topical as well as systemic corticosteroid treatment. Here we describe this unusual presentation of SHML to inform physicians of the possibility for this systemic granulomatous disease to cotribute to relapsing uveitis. |
本系統中英文摘要資訊取自各篇刊載內容。