Mixed-type Multicentric Castleman's Disease Developing during a 17-year Follow-up of Sarcoidosis

Awano, Nobuyasu; Inomata, Minoru; Kondoh, Keisuke; Satake, Kohta; Kamiya, Hiroyuki; Moriya, Atsuko; Ando, Tsunehiro; Kumasaka, Takashi; Takemura, Tamiko; Takeuchi, Kengo; Ikushima, Soichiro

doi:10.2169/internalmedicine.51.8120

Cited by 14 publications

(8 citation statements)

References 16 publications

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“…Another case of MCD accompanied by ascites, pleural effusion and thrombocytopenia was recently reported by Awano et al (12). As observed in our case, the serum IL6 level was only slightly elevated despite a marked increase in the serum level of CRP.…”

Section: Discussionsupporting

confidence: 75%

See 1 more Smart Citation

Successful Treatment of a Patient with Multicentric Castleman's Disease who Presented with Thrombocytopenia, Ascites, Renal Failure and Myelofibrosis Using Tocilizumab, an Anti-Interleukin-6 Receptor Antibody

et al. 2013

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We herein describe an unusual case of multicentric Castleman's disease accompanied by thrombocytopenia, ascites, renal failure and myelofibrosis in a Japanese woman. The patient was initially diagnosed as having myelodysplastic syndrome with myelofibrosis. The general condition of the patient deteriorated rapidly; however, treatment with tocilizumab, an anti-interleukin-6 receptor antibody, together with corticosteroids dramatically improved her symptoms. The clinical features of this case were similar to those of three cases previously reported by Takai et al. (Rinsho Ketsueki, 2010, 51:320-5), which were determined to be thrombocytopenia, anasarca, fever, reticulin myelofibrosis and organomegaly (TAFRO) syndrome, a possibly distinct clinical entity.

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Section: Discussionsupporting

confidence: 75%

“…The administra-tion of tocilizumab dramatically improved the patient's symptoms, as in our case. Therefore, fever, lymphadenopathy with the histopathological features of CD, thrombocytopenia and ascites and/or pleural effusion are features that were common to the current case and the cases reported by Kojima et al (10) and Awano et al (12).…”

Section: Discussionsupporting

confidence: 73%

Successful Treatment of a Patient with Multicentric Castleman's Disease who Presented with Thrombocytopenia, Ascites, Renal Failure and Myelofibrosis Using Tocilizumab, an Anti-Interleukin-6 Receptor Antibody

et al. 2013

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“…The complication of pulmonary sarcoidosis and MCD is rare. Except for our patient, only two cases have been reported as complications or simultaneous occurrences of MCD with pulmonary sarcoidosis . The relationship between these two diseases is not fully understood.…”

Section: Discussionmentioning

confidence: 68%

Multicentric Castleman's disease developing during follow‐up of sarcoidosis

Sawata

Bando

Nakayama

et al. 2016

Respirology Case Reports

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Pulmonary sarcoidosis is reported to have complication of lymphoproliferative disease such as malignant lymphoma, but the complication of multicentric Castleman's disease (MCD) is rarely reported. In our case of a 60‐year‐old woman, bilateral hilar lymphadenopathy was noted in her chest X‐ray. We performed a transbronchial lung biopsy. She was diagnosed as having pulmonary sarcoidosis (Stage II). The shadow on chest X‐ray disappeared without treatment. However, after 8 years, swelling of the mediastinal and abdominal lymph node, thickened bronchovascular bundle, and multiple nodular shadows were identified, and a thoracoscopic lung biopsy was performed. Based on the histopathological findings and elevated serum interleukin‐6 level (75.7 pg/mL), she was diagnosed with pulmonary sarcoidosis complicated by MCD. When a change in chest X‐ray findings are found during monitoring of pulmonary sarcoidosis, it is important to proceed with a thoracoscopic lung biopsy, because of the possibility of the rare complication of MCD.

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“…Very few reports document an association between sarcoidosis and CD [4,5], Crohns disease and CD [6,7], and CD with other gastrointestinal autoimmune disorders such as celiac disease [8] and ulcerative colitis [9]. Immune dysregulation (autoimmunity and immune deficiency), genetic and environmental factors including infectious agents may play roles in pathogenesis of these three disorders, sarcoidosis, Crohn disease and CD.…”

Section: Discussionmentioning

confidence: 99%