Department of Nephrology, Kansai Electric Power Hospital, Japan.
Department of Rheumatology, Kansai Electric Power Hospital, Japan.
Intern Med. 2022 Apr 1;61(7):1027-1032. doi: 10.2169/internalmedicine.8046-21. Epub 2021 Sep 18.
Rosai-Dorfman-Destombes disease (RDD) is a non-Langerhans cell histiocytosis characterized by the accumulation of histiocytes inside the lymph nodes or extranodally. The association between RDD and IgG4-related disease (IgG4-RD) is discussed. We herein report a case of RDD manifesting as acute tubulointerstitial nephritis mimicking IgG4-RD. The first renal biopsy showed severe tubulointerstitial nephritis with infiltration of S100-positive histiocytes and IgG4-positive plasma cells; storiform fibrosis and obliterative phlebitis were not confirmed. After prednisolone therapy, IgG4-positive cells and S100-positive histiocytes were decreased, but the IgG4/IgG ratio increased despite clinical improvement. These findings indicated extranodal RDD in the kidney presenting as tubulointerstitial nephritis.
罗-道-德斯病(RDD)是一种非朗格汉斯细胞组织细胞增生症,其特征是组织细胞在淋巴结内或淋巴结外积聚。本文探讨了 RDD 与 IgG4 相关疾病(IgG4-RD)之间的关系。本文报告了一例表现为急性肾小管间质性肾炎、类似于 IgG4-RD 的 RDD 病例。首次肾活检显示严重的肾小管间质性肾炎,有 S100 阳性组织细胞和 IgG4 阳性浆细胞浸润;未证实存在席纹状纤维化和闭塞性静脉炎。泼尼松龙治疗后,IgG4 阳性细胞和 S100 阳性组织细胞减少,但尽管临床改善,IgG4/IgG 比值增加。这些发现表明肾脏的结外 RDD 表现为肾小管间质性肾炎。