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1. Sugimoto T, Ito J, Takeda N, Gasyu I, Okazaki T, Sakaguchi M, Osawa N, Tanaka Y, Oka K, Uzu T, Kashiwagi A: A case of Castleman's disease complicated with nephrotic syndrome due to glomerulopathy mimicking membranoproliferative glomerulonephritis. Am J Med Sci; 2008 Jun;335(6):495-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] A case of Castleman's disease complicated with nephrotic syndrome due to glomerulopathy mimicking membranoproliferative glomerulonephritis.
  • Castleman's disease is a rare atypical lymphoproliferative disorder.
  • Renal manifestations, such as proteinuria, hematuria, and renal dysfunction, are common in Castleman's disease; however, a nephrotic syndrome rarely occurs.
  • We have encountered an unusual case of Castleman's disease of the plasma cell type characterized by nephrotic syndrome because of glomerulopathy mimicking membranoproliferative glomerulonephritis.
  • Our patient showed higher levels of circulating cytokines (interleukin-6/vascular endothelial cell-derived growth factor), the glomerular lesions not associated with immunocomplex deposition, and the resolution of nephrotic syndrome after successful corticosteroids therapy resulting in a decline in cytokines levels, thereby implicating a cytokine-induced glomerular cell injury/activation as a possible cause of the glomerular pathological changes in this case.
  • [MeSH-major] Giant Lymph Node Hyperplasia / complications. Glomerulonephritis, Membranoproliferative / pathology. Kidney Glomerulus / ultrastructure. Nephrotic Syndrome / complications
  • [MeSH-minor] Adrenal Cortex Hormones / therapeutic use. Diagnosis, Differential. Female. Glomerulonephritis / complications. Glomerulonephritis / drug therapy. Glomerulonephritis / pathology. Humans. Middle Aged. Prednisolone / therapeutic use. Treatment Outcome

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  • (PMID = 18552583.001).
  • [ISSN] 0002-9629
  • [Journal-full-title] The American journal of the medical sciences
  • [ISO-abbreviation] Am. J. Med. Sci.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Adrenal Cortex Hormones; 9PHQ9Y1OLM / Prednisolone
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2. Viejo-Borbolla A, Schulz TF: Kaposi's sarcoma-associated herpesvirus (KSHV/HHV8): key aspects of epidemiology and pathogenesis. AIDS Rev; 2003 Oct-Dec;5(4):222-9
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  • The search for a transmissible infectious agent as the cause of Kaposi's sarcoma lead to the discovery in 1994 of Kaposi's sarcoma-associated herpesvirus (KSHV), also known as human herpesvirus type 8 (HHV8).
  • KSHV is the only human gamma2 herpesvirus (rhadinovirus) known so far, and is also associated with two other AIDS-related lymphoproliferative disorders: primary effusion lymphoma (PEL) and the plasma-cell variant of multicentric Castleman's disease (MCD).
  • [MeSH-minor] Giant Lymph Node Hyperplasia / epidemiology. Giant Lymph Node Hyperplasia / virology. Humans. Lymphoma, AIDS-Related / epidemiology. Lymphoma, AIDS-Related / virology. Viral Proteins / genetics. Viral Proteins / metabolism. Virus Latency

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  • (PMID = 15012001.001).
  • [ISSN] 1139-6121
  • [Journal-full-title] AIDS reviews
  • [ISO-abbreviation] AIDS Rev
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Spain
  • [Chemical-registry-number] 0 / Viral Proteins
  • [Number-of-references] 120
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3. McClain KL, Natkunam Y, Swerdlow SH: Atypical cellular disorders. Hematology Am Soc Hematol Educ Program; 2004;:283-96
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  • What are the similarities and the differences between Langerhans cell histiocytosis (LCH), sinus histiocytosis with massive lymphadenopathy (SHML) or Rosai-Dorfman disease, and Castleman's disease (CD)?
  • Advocates of the malignant proliferation theory rest their case primarily on the following two points: Clonality of the CD1a+ Langerhans cells was demonstrated by analysis of the human androgen receptor in patients with single bone lesions (Low Risk) or multisystem disease including spleen, liver, bone marrow, or lung (High Risk).
  • The diagnosis rests on finding intact lymphocytes in the cytoplasm of activated macrophages as well as accumulation of mature plasma cells.
  • An intriguing finding of human herpesvirus (HHV)-6 viral proteins in SHML has been reported in several patients, but needs further study.
  • Localized cases are divided into the hyaline vascular type and plasma cell type.
  • The two types are distinguished largely by the nature of the follicles and the number of interfollicular plasma cells.
  • Interleukin (IL)-6 expression is increased in the plasma cell type.
  • Multicentric CD of the plasmablastic type is most often found in HIV-positive patients with coincident HHV-8 infection.
  • Other cases of multicentric CD are also most like the plasma cell type, however, with disseminated disease and constitutional symptoms.
  • A wide variety of anti-neoplastic drugs, radiation therapy, anti-IL-6 and rituximab or atlizumab have been used with varying success in patients with multicentric CD.

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  • (PMID = 15561688.001).
  • [ISSN] 1520-4391
  • [Journal-full-title] Hematology. American Society of Hematology. Education Program
  • [ISO-abbreviation] Hematology Am Soc Hematol Educ Program
  • [Language] ENG
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 77
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4. Carbone A, Cesarman E, Spina M, Gloghini A, Schulz TF: HIV-associated lymphomas and gamma-herpesviruses. Blood; 2009 Feb 5;113(6):1213-24
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  • Among the most common HIV-associated lymphomas are Burkitt lymphoma (BL) and diffuse large B-cell lymphoma (DLBCL) with immunoblastic-plasmacytoid differentiation (also involving the central nervous system).
  • Lymphomas occurring specifically in HIV-positive patients include primary effusion lymphoma (PEL) and its solid variants, plasmablastic lymphoma of the oral cavity type and large B-cell lymphoma arising in Kaposi sarcoma herpesvirus (KSHV)-associated multicentric Castleman disease.
  • These lymphomas together with BL and DLBCL with immunoblastic-plasmacytoid differentiation frequently carry EBV infection and display a phenotype related to plasma cells.
  • (2) AIDS lymphomas fall in a spectrum of B-cell differentiation where those associated with EBV or KSHV commonly exhibit plasmablastic differentiation; and (3) prognosis for patients with lymphomas and concomitant HIV infection could be improved using better combined chemotherapy protocols incorporating anticancer treatments and antiretroviral drugs.
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Antiviral Agents / therapeutic use. Humans. Tumor Virus Infections / pathology. Tumor Virus Infections / therapy. Tumor Virus Infections / virology


5. Yu GP, Wang SJ, Shi J, Zhao YQ: [Clinical features and prognosis in 49 patients with Castleman's disease]. Zhongguo Yi Xue Ke Xue Yuan Xue Bao; 2009 Oct;31(5):570-4
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Clinical features and prognosis in 49 patients with Castleman's disease].
  • OBJECTIVE: To analyze the clinical features and prognosis of patients with Castleman's disease (CD).
  • RESULTS: In patients with uni-centric CD (UCD), hyaline vascular type had the highest percentage (88.2%, 15/17), which was significantly higher than that of either plasma cell type (5.9%, 1/17) or mixed cell type (5.9%, 1/17) (P < 0.05).
  • Twenty-three out of 29 patients with MCD were treated with chemotherapy; only 6 patients achieved CR and 9 achieved partial remission (PR), and the overall response rate was 65.2%.
  • New drugs and clinical trials are needed to improve the outcome of MCD.
  • [MeSH-major] Giant Lymph Node Hyperplasia / diagnosis. Giant Lymph Node Hyperplasia / therapy

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  • (PMID = 19968073.001).
  • [ISSN] 1000-503X
  • [Journal-full-title] Zhongguo yi xue ke xue yuan xue bao. Acta Academiae Medicinae Sinicae
  • [ISO-abbreviation] Zhongguo Yi Xue Ke Xue Yuan Xue Bao
  • [Language] chi
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] China
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6. Miltenyi Z, Toth J, Gonda A, Tar I, Remenyik E, Illes A: Successful immunomodulatory therapy in castleman disease with paraneoplastic pemphigus vulgaris. Pathol Oncol Res; 2009 Sep;15(3):375-81
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  • [Title] Successful immunomodulatory therapy in castleman disease with paraneoplastic pemphigus vulgaris.
  • Castleman disease is a rare lymphoproliferative disorder.
  • The clinical signs and symptoms of the disease are primarily mediated by cytokines, especially interleukin-6.
  • Search for neoplasm revealed a retroperitoneal Castleman tumour sized 15 x 6 x 5 cm in the abdominal MRI.
  • Histological examination showed hyalin hypervascular Castleman disease.
  • Considering her young, fertile age and the multicentric Castleman disease, non-cytostatic immunomodulatory therapy was started including steroid, cyclosporine-A and thalidomide treatment.
  • At this time the histological examination showed transient type tumour between plasma cell and vascular variant.
  • 4 years after the onset of the disease. (18)FDG PET/CT examination showed low metabolic active mass in the right iliacal region, but our patient had no symptoms or complaints.
  • Castleman disease can be successfully treated with non-cytostatic immunomodulatory therapy.
  • [MeSH-major] Giant Lymph Node Hyperplasia / drug therapy. Immunomodulation. Immunosuppressive Agents / therapeutic use. Paraneoplastic Syndromes / drug therapy. Pemphigus / drug therapy
  • [MeSH-minor] Adrenal Cortex Hormones / therapeutic use. Cyclosporine / therapeutic use. Female. Humans. Neoplasm, Residual / drug therapy. Neoplasm, Residual / pathology. Retroperitoneal Neoplasms / complications. Retroperitoneal Neoplasms / drug therapy. Retroperitoneal Neoplasms / pathology. Thalidomide / therapeutic use

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  • (PMID = 19067241.001).
  • [ISSN] 1532-2807
  • [Journal-full-title] Pathology oncology research : POR
  • [ISO-abbreviation] Pathol. Oncol. Res.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Netherlands
  • [Chemical-registry-number] 0 / Adrenal Cortex Hormones; 0 / Immunosuppressive Agents; 4Z8R6ORS6L / Thalidomide; 83HN0GTJ6D / Cyclosporine
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7. Gholam D, Vantelon JM, Al-Jijakli A, Bourhis JH: A case of multicentric Castleman's disease associated with advanced systemic amyloidosis treated with chemotherapy and anti-CD20 monoclonal antibody. Ann Hematol; 2003 Dec;82(12):766-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] A case of multicentric Castleman's disease associated with advanced systemic amyloidosis treated with chemotherapy and anti-CD20 monoclonal antibody.
  • Multicentric Castleman's disease (MCD) is a rare systemic lymphoproliferative disorder with too few patient series reported in the literature to have a clear idea about the etiology, outcome and the best treatment available.
  • We report a case of a 28-year-old patient with plasma-cell type, human immunodeficiency virus (HIV)-negative and human herpesvirus-8 (HHV-8)-negative MCD who responded to treatment with chemotherapy and the anti-CD20 monoclonal antibody, rituximab.
  • [MeSH-major] Amyloidosis / complications. Amyloidosis / drug therapy. Antibodies, Monoclonal / administration & dosage. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Giant Lymph Node Hyperplasia / complications. Giant Lymph Node Hyperplasia / drug therapy
  • [MeSH-minor] Adult. Antibodies, Monoclonal, Murine-Derived. Female. Humans. Multiple Organ Failure / drug therapy. Rituximab


8. Minami R, Yamamoto M: [A case of HIV-1 and HHV-8-associated Castleman disease with a relapsing high fever and lymphoadenopathy]. Kansenshogaku Zasshi; 2006 Jul;80(4):423-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [A case of HIV-1 and HHV-8-associated Castleman disease with a relapsing high fever and lymphoadenopathy].
  • Examination of a cervical lymph node specimen resulted in a diagnosis of Castleman disease, plasma cell type.
  • Since the plasma HHV-8 DNA and serum IL-6 were elevated during the flare-up, were negative between episodes, he was treated with ART to control the Castleman disease.
  • Castleman disease is an unusual complication in patients with HIV-1 and HHV-8 infection, but it should be included in the differential diagnosis of patients who exhibit a relapsing systemic inflammatory syndrome and lymphoadenopathy.
  • Further study is needed to determine the appropriate usage and timing of the anti-HHV-8 and HIV-1 medication.
  • [MeSH-major] Giant Lymph Node Hyperplasia / etiology. HIV Infections / complications. HIV-1. Herpesviridae Infections / complications. Herpesvirus 8, Human. Lymphatic Diseases / complications. Relapsing Fever / complications


9. Al-Maghrabi J, Kamel-Reid S, Bailey DJ: Lack of evidence of Epstein-Barr virus infection in patients with Castleman's disease. Molecular genetic analysis. Saudi Med J; 2006 Aug;27(8):1125-9
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  • [Title] Lack of evidence of Epstein-Barr virus infection in patients with Castleman's disease. Molecular genetic analysis.
  • Castleman's disease (CD) is atypical lymphoproliferative disorder.
  • METHODS: We searched medical records for cases of CD at the Toronto General Hospital, Toronto, Canada and King Abdulaziz University Hospital, Jeddah, Saudi Arabia.
  • Polymerase chain reaction were performed at the Department of Pathology and Laboratory Medicine, Toronto General Hospital.
  • Epstein-Barr virus genome was detected only in 2 cases; both were males and have plasma cell type.
  • One is a localized type and the other is of a multicentric type.
  • [MeSH-major] Giant Lymph Node Hyperplasia / virology. Herpesviridae Infections. Herpesvirus 4, Human / isolation & purification. Tumor Virus Infections

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  • (PMID = 16883438.001).
  • [ISSN] 0379-5284
  • [Journal-full-title] Saudi medical journal
  • [ISO-abbreviation] Saudi Med J
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Saudi Arabia
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10. Al-Maghrabi JA, Kamel-Reid S, Bailey DJ: Lack of evidence of Epstein-Barr virus infection in patients with Castleman`s disease. Molecular genetic analysis. Neurosciences (Riyadh); 2006 Oct;11(4):279-83
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Lack of evidence of Epstein-Barr virus infection in patients with Castleman`s disease. Molecular genetic analysis.
  • Castleman`s disease (CD) is atypical lymphoproliferative disorder.
  • METHODS: We searched medical records for cases of CD at the Toronto General Hospital, Toronto, Canada and King Abdulaziz University Hospital, Jeddah, Saudi Arabia.
  • Polymerase chain reaction were performed at the Department of Pathology and Laboratory Medicine, Toronto General Hospital.
  • Epstein-Barr virus genome was detected only in 2 cases; both were males and have plasma cell type.
  • One is a localized type and the other is of a multicentric type.

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  • (PMID = 22266437.001).
  • [ISSN] 1319-6138
  • [Journal-full-title] Neurosciences (Riyadh, Saudi Arabia)
  • [ISO-abbreviation] Neurosciences (Riyadh)
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Saudi Arabia
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11. Nakamichi N, Morii E, Ikeda J, Qiu Y, Mamato S, Tian T, Fukuhara S, Aozasa K: Synergistic effect of interleukin-6 and endoplasmic reticulum stress inducers on the high level of ABCG2 expression in plasma cells. Lab Invest; 2009 Mar;89(3):327-36
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Synergistic effect of interleukin-6 and endoplasmic reticulum stress inducers on the high level of ABCG2 expression in plasma cells.
  • To date, manner of ABCG2 expression in lymphoid tissues is not known.
  • Immunohistochemically, strong ABCG2 expression was found in a small proportion of plasma cells mainly located in the interfollicular space of lymphoid tissues.
  • The number of ABCG2-high plasma cells increased in interleukin-6- (IL-6) rich lesions, such as Castleman's disease of plasma cell type.
  • Plasma cells are subjected to endoplasmic reticulum (ER) stress when excess proteins are synthesized, and IL-6 stimulates protein synthesis.
  • Therefore, the effect of IL-6 and ER stress on ABCG2 expression in plasma cells was examined.
  • Knocked-down of ABCG2 by siRNA or ABCG2 inhibitor reduced plasma cell viability under ER stress.
  • These suggest that ABCG2 is a surviving factor for plasma cells.
  • [MeSH-major] ATP-Binding Cassette Transporters / metabolism. Endoplasmic Reticulum / metabolism. Interleukin-6 / metabolism. Neoplasm Proteins / metabolism. Plasma Cells / metabolism. Stress, Physiological
  • [MeSH-minor] ATP Binding Cassette Transporter, Sub-Family G, Member 2. Apoptosis / drug effects. Caspase 3 / metabolism. Cell Line. DNA Methylation / drug effects. Gene Expression. Humans. Immunohistochemistry. Lymph Nodes / metabolism. Lymph Nodes / pathology. Lymphoma / metabolism. Palatine Tonsil / metabolism. Palatine Tonsil / pathology. Promoter Regions, Genetic. Thapsigargin / pharmacology. Tunicamycin / pharmacology

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  • (PMID = 19139722.001).
  • [ISSN] 1530-0307
  • [Journal-full-title] Laboratory investigation; a journal of technical methods and pathology
  • [ISO-abbreviation] Lab. Invest.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / ABCG2 protein, human; 0 / ATP Binding Cassette Transporter, Sub-Family G, Member 2; 0 / ATP-Binding Cassette Transporters; 0 / Interleukin-6; 0 / Neoplasm Proteins; 11089-65-9 / Tunicamycin; 67526-95-8 / Thapsigargin; EC 3.4.22.- / Caspase 3
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12. Chen CH, Liu HC, Hung TT, Liu TP: Possible roles of Epstein-Barr virus in Castleman disease. J Cardiothorac Surg; 2009;4:31
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Possible roles of Epstein-Barr virus in Castleman disease.
  • BACKGROUND: Complete resection seemed to be curative in patients with Castleman disease of any location but the disease is likely to be reactive in its pathogenesis.
  • The relation between Epstein-Barr virus and Castleman disease has not been elucidated.
  • We tried to define the role of Epstein-Barr virus in the pathogenesis of Castleman disease.
  • METHODS: 20 cases of Castleman disease were retrospectively reviewed from 1993 to 2006.
  • At least 2 to 4 representative sections of formalin-fixed, paraffin-embedded specimens from each patient were obtained to examine the presence of EBV and its localization by hematoxylin-eosin stain, immunohistochemistry, polymerase chain reaction and In-situ hybridization RESULTS: Hyaline-vascular type was diagnosed in 18 cases, plasma cell type in 1 and mixed type in 1 case.
  • CONCLUSION: There is a strong association between Castleman disease and Epstein-Barr virus.
  • EBV may have a potential role in angiogenesis of Castleman disease.
  • For smaller lesion with high activity of angiogenesis but not amenable for curative resection, anti-angiogenesis medications may have a potential role to control the disease.
  • [MeSH-major] Epstein-Barr Virus Infections / complications. Giant Lymph Node Hyperplasia / virology

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  • (PMID = 19589162.001).
  • [ISSN] 1749-8090
  • [Journal-full-title] Journal of cardiothoracic surgery
  • [ISO-abbreviation] J Cardiothorac Surg
  • [Language] eng
  • [Publication-type] Journal Article
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13. Al Otaibi T, Al Sagheir A, Ludwin D, Meyer R: Post renal transplant Castleman's disease resolved after graft nephrectomy: a case report. Transplant Proc; 2007 May;39(4):1276-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Post renal transplant Castleman's disease resolved after graft nephrectomy: a case report.
  • Angiofollicular lymphoid hyperplasia (Castleman's disease) is a lymphoproliferative process thought to be mediated by overexpression of II interleukin-6.
  • Castleman's disease has two variants: Castleman's disease has two variants: Hyaline vascular type and plasma cell variant (multicentric Castleman's disease).
  • The hyaline vascular type tends to be localized, and the plasma cell variant shows more systematic signs and carriers a worse clinical prognosis.
  • Castleman's disease is associated with B-cell lymphoma, Kaposi sarcoma, Human herpes virus 8 (HHV-8), and Epstein-Barr virus.
  • Castleman's disease have been described thrice post kidney transplant.
  • In this report, we document the course of a renal recipient who developed the plasma cell variant of Castleman's disease at 16 months after failure of his allograft and return to dialysis.
  • To our knowledge, this is the first case where the disease manifestations disappeared after graft removal.
  • Our patient experienced chronic renal allograft rejection which may have driven all the systematic manifestations of multicentric castleman's disease and possibly reactivated a latent HHV-8 infection.
  • In this case immunohistochemical testing for HHV-8 was not available to prove a role for this agent.
  • [MeSH-major] Giant Lymph Node Hyperplasia / diagnosis. Kidney Transplantation / adverse effects
  • [MeSH-minor] Humans. Immunosuppressive Agents / therapeutic use. Lymph Nodes / pathology. Male. Middle Aged. Nephrectomy. Renal Dialysis. Reoperation


14. Kojima M, Nakamura S, Nishikawa M, Itoh H, Miyawaki S, Masawa N: Idiopathic multicentric Castleman's disease. A clinicopathologic and immunohistochemical study of five cases. Pathol Res Pract; 2005;201(4):325-32
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Idiopathic multicentric Castleman's disease. A clinicopathologic and immunohistochemical study of five cases.
  • Several lymphoproliferative disorders may be interpreted as multicentric Castleman's disease (MCD) clinicopathologically.
  • These include HIV infection, autoimmune-disease-associated lymphadenopathy, idiopathic plasmacytic lymphadenopathy with polyclonal hyperimmunoglobulinemia, "idiopathic MCD", POEMS syndrome (polyneuropathy, anasarca, organomegaly, endocrinopathy, M-proteins, and skin lesions), and non-Hodgkin's lymphomas.
  • We report on the clinicopathologic and immunohistologic findings of five cases of idiopathic MCD and discuss the problems regarding their differential diagnosis.
  • However, during the course of disease, minimal diagnostic criteria for POEMS syndrome, i.e., monoclonal plasma cell proliferation and sensory motor neuropathys, were absent in all five cases.
  • However, none of them fulfilled the diagnostic criteria for any of the definite autoimmune-disease.
  • Histologically, three lesions exhibited a mixed type of Castleman's disease, and two exhibited the hyaline-vascular type.
  • The majority of the germinal centers were of the hyaline-vascular or epithelioid germinal type, with a few hyperplastic germinal centers.
  • Moderate to large sheets of plasma cells were observed in three mixed type cases.
  • [MeSH-major] Germinal Center / pathology. Giant Lymph Node Hyperplasia / pathology. Lymph Nodes / pathology
  • [MeSH-minor] Aged. Biomarkers / metabolism. Diagnosis, Differential. Drug Therapy, Combination. Female. Humans. Hyperplasia. Immunoenzyme Techniques. Male. Middle Aged. POEMS Syndrome / diagnosis. Treatment Outcome






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