Im Jeong Soo, Yoon Hyun Jung, Kim Hae Su, Lee Miji
J Korean Soc Radiol. 2025 Mar;86(2):291-297. doi: 10.3348/jksr.2024.0078. Epub 2025 Mar 26.
Rosai-Dorfman disease (RDD) is an uncommon proliferative histiocytic disorder of unknown etiology typically presenting as massive lymphadenopathy, and in some cases, with extranodal involvement. However, serous membranes are rarely involved in extranodal RDDs, and reports regarding pericardial involvement are scarce. Herein, we report a case of extranodal RDD manifesting as diffuse pericardial thickening and effusion in a 79-year-old man with monoclonal gammopathy. The patient complained of dyspnea, hence chest CT and PET scans were performed. They showed irregular thickening of the pericardium with a marked increase in metabolic activity. Pericardial biopsy showed the characteristic S100-positive and CD68-positive histiocytes exhibiting emperipolesis. The disease progressively evolved to bilateral pleural thickening with effusion of the pericardium, and finally led to death even with corticosteroid treatment. Although exceedingly rare, this case demonstrates the importance of RDD in the differential diagnosis of effusion in serous cavities based on imaging findings.
罗萨伊-多夫曼病(RDD)是一种病因不明的罕见增殖性组织细胞疾病,通常表现为巨大淋巴结病,在某些情况下伴有结外受累。然而,浆膜很少累及结外RDD,关于心包受累的报道也很少。在此,我们报告一例79岁患有单克隆丙种球蛋白病的男性,其结外RDD表现为弥漫性心包增厚和积液。患者主诉呼吸困难,因此进行了胸部CT和PET扫描。结果显示心包不规则增厚,代谢活性显著增加。心包活检显示特征性的S100阳性和CD68阳性组织细胞呈现吞噬现象。该疾病逐渐发展为双侧胸膜增厚并伴有心包积液,即使接受皮质类固醇治疗最终仍导致死亡。尽管极为罕见,但该病例证明了RDD在基于影像学表现对浆膜腔积液进行鉴别诊断中的重要性。