Nabeya Daijiro, Yoshimatsu Yuki, Fujiwara Hiroshi
Department of Respiratory Medicine, Okinawa Chubu Hospital, Okinawa, Japan.
Department of Infectious Diseases, Respiratory, and Digestive Medicine, Graduate School of Medicine, University of the Ryukyus, Okinawa, Japan.
Respir Med Case Rep. 2020 Sep 11;31:101216. doi: 10.1016/j.rmcr.2020.101216. eCollection 2020.
It has been considered that idiopathic multicentric Castleman disease often involves pulmonary complications recognized as lymphocytic interstitial pneumonia. On the other hand, recent reports show that the computed tomography often show diffuse interstitial lung disease inconsistence with lymphocytic interstitial pneumonia. Pulmonary diseases with idiopathic multicentric Castleman disease are still rare and poorly understood. Here, we report a case of acute progressive diffuse interstitial lung disease, diagnosed as non-specific interstitial pneumonia, preceding idiopathic multicentric Castleman disease. A 65-year-old male visited our outpatient clinic for dyspnea on exertion. Imaging tests revealed interstitial lung disease showing non-specific interstitial pneumonia pattern, pulmonary function test proved the decline of vital capacity and laboratory tests showed increased fibrosis biomarkers; therefore, initially, he had been diagnosed as non-specific interstitial pneumonia. However, imaging tests also showed mediastinum lymphadenopathy, and laboratory tests revealed increased interleukin-6. Idiopathic multicentric Castleman disease was suspected. The lung and mediastinum lymph node biopsies were performed, and pathological findings of the lymph nodes were compatible with multicentric Castleman disease. Pathological findings of the lung showed that the fibrous thickening of interstitium and the collapse of alveoli. We diagnosed this case as idiopathic multicentric Castleman disease preceded by diffuse interstitial lung disease. Treatment with prednisolone improved the dyspnea, and the pulmonary lesions disappeared. The presented case suggests that interstitial lung disease could precede idiopathic multicentric Castleman disease. Chest physicians should be aware that idiopathic multicentric Castleman disease is one of the causative diseases of diffuse interstitial lung disease like non-specific interstitial pneumonia on the chest images.
人们认为,特发性多中心Castleman病常伴有被认为是淋巴细胞间质性肺炎的肺部并发症。另一方面,最近的报告显示,计算机断层扫描常显示与淋巴细胞间质性肺炎不一致的弥漫性间质性肺疾病。特发性多中心Castleman病相关的肺部疾病仍然罕见且了解甚少。在此,我们报告一例急性进行性弥漫性间质性肺疾病病例,在特发性多中心Castleman病之前被诊断为非特异性间质性肺炎。一名65岁男性因劳力性呼吸困难前来我院门诊就诊。影像学检查显示间质性肺疾病呈非特异性间质性肺炎模式,肺功能测试证明肺活量下降,实验室检查显示纤维化生物标志物升高;因此,最初他被诊断为非特异性间质性肺炎。然而,影像学检查还显示纵隔淋巴结肿大,实验室检查显示白细胞介素-6升高。怀疑为特发性多中心Castleman病。进行了肺和纵隔淋巴结活检,淋巴结的病理结果与多中心Castleman病相符。肺的病理结果显示间质纤维增厚和肺泡塌陷。我们将此病例诊断为弥漫性间质性肺疾病在先的特发性多中心Castleman病。泼尼松龙治疗改善了呼吸困难,肺部病变消失。该病例提示间质性肺疾病可能先于特发性多中心Castleman病出现。胸科医生应意识到,特发性多中心Castleman病是胸部影像上如非特异性间质性肺炎等弥漫性间质性肺疾病的病因之一。