Singh Neetu, Sharma Ramesh, Verma Umesh P, Singh Sanjay K, Dixit Jaya, Balapure Anil K
Division of Toxicology, Central Drug Research Institute, M.G. Marg, Lucknow-226001, UP, India.
Ear Nose Throat J. 2009 Mar;88(3):E20-3.
Rosai-Dorfman disease is rare. When it does occur, it usually affects children, and it has a propensity for whites and blacks as opposed to members of other races. We report the case of a 45-year-old Asian Indian woman who presented with painful masses in the area of the axillary, cervical, and inguinal lymph nodes. She had a decade-long history of tonic-clonic seizures and a recent history of digestive complaints and progressive nasal stuffiness. Endoscopic examination of the left nasal cavity revealed the presence of submucosal bulges along the septum and the lateral wall; hypertrophy of the adenoids was also noted. Histologic analysis of lymph node specimens revealed dilated parenchymal sinuses, germinal activity with infiltration of numerous histiocytes (emperipolesis), and chronic inflammatory cells. The patient was diagnosed with Rosai-Dorfman disease (sinus histiocytosis with massive lymphadenopathy) with atypical extranodal involvement (the nasal area). An aggressive course of polychemotherapy thrice daily was initiated, but it had no lasting effect, and the patient died 8 months later of an undetermined cause. Our patient's age at the onset of her disease, her race, and the extranodal involvement make this case of Rosai-Dorfman disease unusual and perhaps unique.
罗萨伊-多夫曼病较为罕见。发病时通常累及儿童,且相较于其他种族成员,白人和黑人更易患病。我们报告一例45岁的亚洲印度女性病例,该患者腋窝、颈部及腹股沟淋巴结区域出现疼痛性肿块。她有长达十年的强直阵挛性癫痫病史,近期有消化系统不适及进行性鼻塞病史。对左侧鼻腔进行内镜检查发现鼻中隔及侧壁有黏膜下隆起;还发现腺样体肥大。淋巴结标本的组织学分析显示实质窦扩张、生发活动伴有大量组织细胞浸润(血细胞吞噬现象)以及慢性炎性细胞。该患者被诊断为罗萨伊-多夫曼病(伴巨大淋巴结病的窦组织细胞增生症),伴有非典型结外受累(鼻腔区域)。开始每日三次进行强化多药化疗,但无持久疗效,患者8个月后因不明原因死亡。我们患者的发病年龄、种族以及结外受累情况使该例罗萨伊-多夫曼病病例不同寻常,甚至可能独一无二。