以淚腺腫瘤表現的羅塞－朵夫曼氏症－病例報告

Rosai-Dorfman Disease Manifested as Lacrimal Gland Tumors-A Case Report

蔡守恒(Shou-Heng Tsai) ； 高啓祥(Shine C. S. Kao) ； 林智一(Tze-Yi Lin)

《中華民國眼科醫學會雜誌》 48卷2期 (2009/06) Pp. 228-233

https://doi.org/10.30048/ACTASOS.200906.0018

摘要

目的：報告一個以淚腺腫瘤為主要表現的羅塞－朵夫曼氏症的病例。
方法：病例報告。
結果：一個58歲男性，本身具有雙眼甲狀腺眼疾。左眼上眼瞼腫脹約半年，在初步懷疑原因不明的眼窩淚腺發炎診斷下，使用了口服類固醇和抗高甲狀腺藥物治療。經過一年後，病人雙眼淚腺腫脹變的更為明顯，於是給予病人口服化學藥物治療，病人雙眼上眼瞼依然腫脹時好時壞。經過三年門診追蹤後，病人接受左眼淚腺診斷式切片手術(surgical incision & biopsy)，病理組織報告確定為羅塞－朵夫曼氏症(Rosai-Dorfman Disease)。在術後門診追蹤，病患並沒有傷口腫脹且復元良好。術後一年的門診觀察，病人雙眼狀況穩定沒有惡化的情況。
結論：羅塞－朵夫曼氏症表現在以雙眼淚腺腫脹為表現症狀的甲狀腺眼疾病人相當地罕見。我們提供了本例此病的病理診斷報告和臨床表現，對於雙眼淚腺腫脹的甲狀腺眼疾病人建議納入鑑別診斷之一。

並列摘要

Purpose: To report a case of Rosai-Dorfman disease manifested as lacrimal gland tumors
Methods: Case report.
Results: A 58-year-old man with hyperthyroidism complained of a left upper eyelid mass for half a year. Idiopathic dacryoadenitis was suspected at first and oral steroid treatment was tried. However, after one year of treatment, the patient's symptoms progressed and he developed intermittent swollen upper eyelids with a palpable mass in both lacrimal gland regions. After three years of OPD follow-up, he finally accepted an incision biopsy of the left lacrimal gland, which showed evidence of Rosai-Dorfman disease in the lacrimal gland. No progression of the disease was noted one year after the surgery.
Conclusions: The histopathology we provided suggests even rare Rosai-Dorfman disease could be one possibility in the differential diagnosis of bilateral lacrimal gland enlargement in a patient with thyroid eye disease.

並列關鍵字

Rosai-Dorfman Disease ； Lacrimal Gland Tumor ； Emperipolesis ； Sinus histiocytosis with massive lymphadenopathy