Harada Naoko, Sayama Koichi, Tanaka Kyuto, Hasegawa Naoki, Okamoto Shinichiro, Hayashi Yuichiro, Ishizaka Akitoshi
Division of Pulmonary Medicine, Department of Internal Medicine, School of Medicine, Keio University.
Nihon Kokyuki Gakkai Zasshi. 2010 Feb;48(2):145-50.
A 46-year-old man was admitted for the evaluation of a dry cough and dyspnea on exertion. Laboratory tests revealed anemia, elevated CRP, polyclonal hyperimmunoglobulinemia, and an elevated interleukin-6 level. Radiological examination of the chest showed peribronchovascular consolidations, ground glass opacities, small nodular opacities, and interlobular septal thickenings in the lungs, accompanied with hilar and mediastinal lymphadenopathies. A video-assisted thoracoscopic lung and mediastinal lymph node biopsy revealed plasmacytic and lymphocytic infiltration around the bronchovascular bundles of the lungs, and plasmacytic infiltration in the interfollicular areas of the nodes. Based on these findings, a diagnosis of multicentric Castleman disease was confirmed. The patient received a humanized anti-interleukin-6 receptor antibody, (tocilizumab, 8 mg/kg), every 2 weeks for 3 years, during which time, his PaO2 level improved from 64.1 Torr to 83.4 Torr, vital capacity increased from 2.53 L to 3.95 L, and radiological abnormalities in the lungs gradually improved, suggesting that tocilizumab is effective for interstitial pneumonia in patients with multicentric Castleman disease.
一名46岁男性因劳力性干咳和呼吸困难入院评估。实验室检查显示贫血、CRP升高、多克隆高免疫球蛋白血症以及白细胞介素-6水平升高。胸部影像学检查显示肺部支气管血管周围实变、磨玻璃影、小结节影和小叶间隔增厚,并伴有肺门和纵隔淋巴结肿大。电视辅助胸腔镜肺及纵隔淋巴结活检显示肺支气管血管束周围有浆细胞和淋巴细胞浸润,淋巴结滤泡间区有浆细胞浸润。基于这些发现,确诊为多中心Castleman病。该患者每2周接受一次人源化抗白细胞介素-6受体抗体(托珠单抗,8 mg/kg)治疗,持续3年,在此期间,他的动脉血氧分压水平从64.1 Torr提高到83.4 Torr,肺活量从2.53 L增加到3.95 L,肺部的影像学异常逐渐改善,提示托珠单抗对多中心Castleman病患者的间质性肺炎有效。