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结节病诊断后发生的特发性多中心Castleman病:一例病例报告及文献复习

Idiopathic multicentric Castleman disease developing after a diagnosis of sarcoidosis: A case report and literature review.

作者信息

Muramatsu Takumi, Kono Masato, Ishige Masaki, Saito Takahiko, Higasa Misato, Nihashi Fumiya, Aono Yuya, Katsumata Mineo, Miwa Hideki, Miki Yoshihiro, Misaki Taro, Otsuki Yoshiro, Hashimoto Dai, Enomoto Noriyuki, Suda Takafumi, Nakamura Hidenori

机构信息

Department of Respiratory Medicine, Seirei Hamamatsu General Hospital, Hamamatsu, 430-8558, Japan.

Department of Nephrology, Seirei Hamamatsu General Hospital, Hamamatsu, 430-8558, Japan.

出版信息

Respir Med Case Rep. 2025 Feb 27;54:102188. doi: 10.1016/j.rmcr.2025.102188. eCollection 2025.

Abstract

A 72-year-old woman presented with an abnormal shadow on chest radiograph. She was histologically diagnosed with sarcoidosis 20 years previously, and prednisolone was initiated 8 years previously. Computed tomography revealed centrilobular micronodules and bronchovascular bundle thickening in both lungs with multicentric lymphadenopathies; multiple pulmonary nodular lesions appeared during prednisolone tapering. Laboratory findings included polyclonal hypergammaglobulinemia and elevated interleukin-6 levels. Surgical lung biopsy revealed marked lymphoplasmacytic infiltration with lymphoid aggregates. The patient tested negative for human herpesvirus-8 and clinically diagnosed with idiopathic multicentric Castleman disease. The coexistence of sarcoidosis and Castleman disease is rare; this case improved with additional tocilizumab treatment.

摘要

一名72岁女性因胸部X线片出现异常阴影就诊。她20年前经组织学诊断为结节病,8年前开始使用泼尼松龙治疗。计算机断层扫描显示双肺小叶中心性微结节和支气管血管束增厚,并伴有多中心淋巴结肿大;在泼尼松龙减量过程中出现了多个肺结节性病变。实验室检查结果包括多克隆高球蛋白血症和白细胞介素-6水平升高。手术肺活检显示有明显的淋巴浆细胞浸润及淋巴样聚集。该患者人类疱疹病毒-8检测呈阴性,临床诊断为特发性多中心Castleman病。结节病和Castleman病共存的情况罕见;该病例经加用托珠单抗治疗后病情改善。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/06dc/11915158/fc72a3b83081/gr1.jpg

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