Localised hyaline vascular type of Castleman's disease mimicking adult-onset Still's disease

Department of Pediatrics, Chang Gung Children's Hospital, Kweishan, Taoyuan, Taiwan.
Clinical Rheumatology (Impact Factor: 1.7). 02/1999; 18(6):485-7. DOI: 10.1007/s100670050143
Source: PubMed


A previously healthy 18-year-old boy presented with daily spiking fever, polyarthritis, and evanescent skin rashes, as well as hepatomegaly and Raynaud's phenomena for 2 months. He was initially diagnosed with adult-onset Still's disease (AOSD). During the period of follow-up, intermittent fever and migratory polyarthritis persisted and an insidiously growing mass over the right axillary region was noted 1 year after the diagnosis of AOSD. Excisional biopsy of the mass revealed a group of lymph nodes with histological features of the hyaline vascular type of Castleman's disease. The patient's symptoms disappeared soon after excision of the lymph nodes. evanescent rash, lymphadenopathy, hepatosplenomegaly and serositis [5]. A clinical picture compatible with the diagnosis of AOSD has not been described in the localised hyaline vascular type of Castleman's disease. We report such a case in an 18-year-old male patient who presented prolonged fever and polyarthritis with an initial diagnosis of AOSD. The diagnosis of hyaline vascular type of Castleman's disease was made 1 year later, when the patient developed an insidiously growing mass over the right axilla.

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    • "The diagnosis of CD in these cases is surprising clinically. On the other hand, due to the histopathological similarities CD may mimic some collagen vascular diseases or in some cases, autoimmune disorders and pathological findings of some reactive lymphoid hyperplasias may mimic CD [15] [16] [17] [18]. So CD may cause some difficulties in some situations in rheumatology clinics. "
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    ABSTRACT: Castleman disease (CD) is an uncommon lymphoproliferative disorder with a unique histopathology. Generally diagnosis is not difficult for an experienced pathologist. However, not usually, some histopathological entities or tissue reactions may mimic CD. Here CD-like reaction due to toxic substance ingestion has been presented.
    European Journal of Internal Medicine 06/2009; 20(3):328-30. DOI:10.1016/j.ejim.2008.07.034 · 2.89 Impact Factor
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    ABSTRACT: Adult onset Still's disease (AOSD) is a rare systemic inflammatory disorder of unknown aetiology, frequently accompanying multiple lymphadenopathy. It often mimics malignant lymphoma, and immunohistochemical and molecular studies are needed for definite diagnosis. To aid in diagnosis and understand the pathogenesis of the disease by clarifying lymph node (LN) pathology in AOSD. Thirteen biopsies (one follow up biopsy) and medical records of 12 patients were reviewed. Immunohistochemistry, polymerase chain reaction for T cell receptor gamma chain (TCRgamma) and immunoglobulin heavy chain gene rearrangement, and Epstein-Barr virus in situ hybridisation were performed. Histologically, LN lesions were classified into four patterns. The most common (six biopsies) showed paracortical hyperplasia, with prominent vascular proliferation, scattered large B/T immunoblasts, and infiltration by reactive lymphocytes and inflammatory cells. In the second pattern (two biopsies), paracortical hyperplasia was accompanied by massive sinus histiocytosis and S-100 positive histiocyte aggregates. The third pattern (three patients) showed an exuberant immunoblastic reaction, in the form of patchy/diffuse infiltration of large T immunoblasts with high mitotic activity, although clonal rearrangement of the TCRgamma gene was not detected. The fourth pattern showed distinct follicular hyperplasia (two cases). One patient with a follow up biopsy showed a pattern change from pronounced follicular hyperplasia to atypical paracortical hyperplasia. AOSD LN lesions show a dynamic histological spectrum, including atypical paracortical hyperplasia, burnt out histiocytic reaction, exuberant immunoblastic reaction, and follicular hyperplasia. During the course of disease, LN reactivity changes and mixed B and T cells are involved in the pathogenesis.
    Journal of Clinical Pathology 11/2004; 57(10):1052-6. DOI:10.1136/jcp.2004.018010 · 2.92 Impact Factor
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    ABSTRACT: Castleman's disease (CD) is rare nonmalignant lymphoproliferative illness, of unknown etiology, clinically characterized by isolated or multiple adenomegalies, associated or not with systemic symptoms such as fever and weight loss. These symptoms can lead to a wrong diagnosis of autoimmune illness and the differential diagnosis must be made through histological examination of involved lymph nodes, which shows, in the case of CD, a pattern of plasma cells with hyaline infiltration. A case of a woman of 24-year-old, who initially presented fever, polyarthritis and skin rash, suggestive adult-onset Still's disease is reported; the histological examination confirmed the diagnosis of Castleman's disease.
    Revista Brasileira de Reumatologia 10/2005; 45(5):331-334. DOI:10.1590/S0482-50042005000500011 · 1.09 Impact Factor
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