A case of idiopathic multicentric Castleman disease with pulmonary hyalinizing granuloma-like multiple pulmonary nodules.

A 41-year-old man presented with chronic cough and chest pain. Laboratory tests revealed anemia, inflammation, hypoalbuminemia, polyclonal hypergammaglobulinemia, and elevated interleukin-6 levels. Computed tomography revealed diffuse bilateral pulmonary nodules and multicentric lymphadenopathy. Histopathology of the pulmonary nodule resembled pulmonary hyalinizing granuloma (PHG), whereas lymph node histopathology was consistent with idiopathic multicentric Castleman disease (iMCD). The patient was diagnosed with iMCD involving PHG-like pulmonary nodules. Little is known about the association between these two diseases, and the present case provides insights regarding the relationship between PHG and iMCD.

Hatano H ，Tsujimoto Y ，Watanabe H ，Tsukada A ，Izumi S ，Nagasaka S ，Igari T ，Suzuki M ，Iikura M ，Hojo M ... -  《-》

Pulmonary hyalinizing granuloma with Castleman's disease.

Atagi S ，Sakatani M ，Akira M ，Yamamoto S ，Ueda E ... -  《INTERNAL MEDICINE》

Amyloidosis secondary to intrapulmonary Castleman disease mimicking pulmonary hyalinizing granuloma-like clinical features: A rare case report.

Wang ST ，Wang QP ，Li J ，Zhang T ，Zhang L ，Mao YY ... -  《-》

A case of acute progressive diffuse interstitial lung disease preceding idiopathic multicentric Castleman disease.

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.

Nabeya D ，Yoshimatsu Y ，Fujiwara H 《Respiratory Medicine Case Reports》

Idiopathic multicentric Castleman disease with pulmonary and cutaneous lesions treated with tocilizumab: A case report.

Human herpes virus-8 (HHV-8)-negative, idiopathic multicentric Castleman disease (iMCD) is a rare and life-threatening disorder driven by proinflammatory cytokines, which is still poorly understood. Pulmonary parenchyma lesion is a rare condition in iMCD, which mainly manifests as lymphocytic interstitial pneumonia and is an indicator of severe iMCD. Cutaneous lesion is also very rare and mainly occurs in Asians. There have been few reports of iMCD patients with both skin and lung parenchyma involvement. We present a Chinese man who complained about a 3-year history of intermittent dry cough and a 2-year history of diffuse reddish-brown maculopapules. Laboratory examination revealed polyclonal hypergammaglobulinemia and hypercytokinemia including interleukin 6. Chest computed tomography revealed small patchy shadows with ground-glass nodules scattered in two lobes and mediastinal lymphadenopathy. The pathological result of the lymph node was consistent with the plasma cell type of Castleman disease. As serum human immunodeficiency virus test and HHV-8 staining of the lymph node were negative, the patient was finally diagnosed with HHV-8 negative iMCD. He was treated with tocilizumab at an intravenous (i.v.) dose of 8 mg/kg every 2 wk combined with methylprednisolone at an i.v. dose of 80 mg/d initially with gradual dose tapering. Partial remission was achieved 9 mo later. iMCD with lung parenchyma and skin involvement is a rare condition that requires clinicians' attention and awareness for early diagnosis.

Han PY ，Chi HH ，Su YT 《World Journal of Clinical Cases》