中国麻风皮肤病杂志 ›› 2022, Vol. 38 ›› Issue (10): 686-689.doi: 10.12144/zgmfskin202210686

• 论著 • 上一篇    下一篇

累及皮肤、眼、骨骼的Rosai-Dorfman病一例并文献复习

廖晓捷1,周桂芝1,刘红1,王晓坤2,张福仁1   

  1. 1山东第一医科大学附属皮肤病医院（山东省皮肤病医院)，山东省皮肤病性病防治研究所，山东济南，250022；2山东第一医科大学附属山东省立医院，山东济南，250012
  • 出版日期:2022-10-15 发布日期:2022-08-22

Rare case of Rosai-Dorfman disease involving skin, eye, and bone: a case report

LIAO Xiaojie1, ZHOU Guizhi1, LIU Hong1, WANG Xiaokun2, ZHANG Furen1   

  1. 1 Shandong Provincial Hospital for Skin Diseases & Shandong Provincial Institute of Dermatology and Venereology, Shandong First Medical University, Jinan 250022, China；2 Shandong Provincial Hospital, Shandong First Medical University, Jinan 250012, China
  • Online:2022-10-15 Published:2022-08-22

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摘要： Rosai-Dorfman病(RDD)可累及皮肤、腮腺、眼睑、肝脏或乳房等，但同时累及皮肤、眼、骨骼的RDD尚未见报道。本文报道一例并复习相关文献。患者,男，41 岁。右侧面部出现麻木感3个月，后于相同部位出现紫红色丘疹1个月，此时无系统受累。组织病理检查：真皮内淋巴组织细胞、浆细胞、巨细胞、中性粒细胞浸润。免疫组化示：组织样细胞S100（＋）、CD68（＋）、CD1a（－），诊断：皮肤Rosai-Dorfman病。患者口服沙利度胺及外用丁酸氢化可的松乳膏和他克莫司软膏治疗2个月后皮损消退，但7个月后皮下组织增生累及眼眶并破坏骨质，至眼科行手术治疗，目前仍在随访中。

关键词: 皮肤Rosai-Dorfman病, 眼眶Rosai-Dorfman Disease, 骨质破坏

Abstract: Rosai-Dorfman disease (RDD) can involve skin, parotid gland, eyelid, liver or breast, but the disease involving skin, eyes and bone at the same time has not been reported. We reported a case of RDD involving skin, eyes and bone at the same time. The patient was a 41-year-old male presented with numbness on the right face for 3 months, followed by purplish-red papules on the same area for 1 month, without systemic involvement at the moment. Histological examination showed infiltration of lymphocytes, plasma cells, giant cells, and neutrophils in the dermis. Immunohistochemistry revealed that the infiltrated cells were positive for S100, CD68 and all cells were negative for CD1a. The diagnosis of cutaneous Rosai-Dorfman disease was made. After 2 months of treatment with oral thalidomide, topical hydrocortisone butyrate cream and tacrolimus ointment, the lesions subsided. But after 7 months, the hyperplasia involved the orbit and destroyed the bone, the patient went to ophthalmology department for surgery and the patient is under followed up.

Key words: cutaneous Rosai-Dorfman disease, orbital Rosai-Dorfman disease, bony destruction