Atypical Hyaline Vascular-Type Castleman's Disease With Thrombocytopenia, Anasarca, Fever, and Systemic Lymphadenopathy
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概要
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Recently, atypical Castleman's disease (CD) was reported in Japan. This disease is considered as TAFRO syndrome or non-idiopathic plasmacytic lymphadenopathy (IPL), a constellation of clinical symptoms, namely, thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly without hyper-γ-globulinemia. Histopathologically, this disease is similar to hyaline vascular (HV)-type CD. Here, we present a 43-year-old Japanese woman meeting the clinical criteria of TAFRO syndrome who was successfully treated with combined corticosteroid therapy. She showed a rapidly progressive course of thrombocytopenia, systemic lymphadenopathy, fever, anasarca, and increase in acute inflammatory proteins without hyper-γ-globulinemia. Lymph node biopsy was performed and revealed HV-type CD without human herpes virus 8 infection, which was clinicopathologically compatible with non-IPL. The association of these atypical features with well-known multicentric Castleman's disease (MCD), namely, HV-type histology with systemic lymphadenopathy, marked thrombocytopenia even with a high level of interleukin-6, and increased acute inflammatory proteins without hyper-γ-globulinemia, suggests that TAFRO syndrome as presented in our case is a novel entity, which may have been diagnosed as MCD in the past. To define this novel entity more clearly and to demonstrate its etiology, further nationwide surveys of this syndrome and MCD are needed. [<I>J Clin Exp Hematopathol 53(</I><I>1) : 87-93,</I> <I>2013</I>]
著者
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KONDO Eisei
Department of Hematology and Chemotherapy, Aichi Cancer Center
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Takata Katsuyoshi
Department Of Pathology Okayama University Graduate School Of Medicine Dentistry And Pharmaceutical
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Nakao Shinji
Cellular Transplantation Biology Department Of Respiratory Medicine Kanazawa University Graduate Sch
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Sato Yasuharu
Department Of Pathology Okayama University Graduate School Of Medical Dentistry And Pharmaceutical S
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Ohno Kyotaro
Department Of Pathology Okayama University Graduate School Of Medicine Dentistry And Pharmaceutical Sciences
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Yoshino Tadashi
Department Of Cytopathology Ayabe City Hospital
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NAKAO Shinji
Cellular Transplantation Biology, Kanazawa University Graduate School of Medical Science
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IWAKI Noriko
Department of Hematology and Oncology, Kanazawa University Hospital
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Yoshino Tadashi
Departments of Pathology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Takeuchi Mai
Departments of Pathology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Orita Yorihisa
Otolaryngology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Yoshino Tadashi
Department of Chemical Engineering, Hiroshima University
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Sato Yasuharu
Departments of Pathology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Kondo Eisei
Departments of Hematology and Oncology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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