scholarly journals A case of Castleman disease with hemophagocytic syndrome derived from HHV8 infection

2021 ◽  
Vol 26 (1) ◽  
Author(s):  
Xiao Cui ◽  
Yongfeng Wu ◽  
Lin Jia ◽  
Jing Chang ◽  
Chuanyun Li ◽  
...  
Keyword(s):  
Hemophagocytic Syndrome ◽  
Genetic Disorders ◽  
Human Herpesvirus ◽  
Castleman Disease ◽  
Metagenomic Sequencing ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Lymphoid Malignancies ◽  
Hhv8 Infection ◽  
Diagnostic Clue

Abstract Background For a patient presenting with fever, multiple lymphadenopathy and splenomegaly, pathogen infection should be preferentially considered, followed by lymphoid malignancies. When traditional laboratory and pathological detection cannot find the pathogenic microorganism, metagenomic sequencing (MGS) which targets the person’s genome for exceptional genetic disorders may detect a rare pathogen. Case presentation Here, we introduced the diagnostic clue of a case of multicentric Castleman disease (MCD) with hemophagocytic syndrome which was elicited from the detection of human herpesvirus-8 in the blood of a HIV-1 infected person by MGS technology during pathogen inspection. This case highlights the need to increase the awareness of MCD among clinicians and pathologists. Conclusions MGS technology may play a pivotal role in providing diagnostic clues during pathogen inspection, especially when pathogens are not detectable by conventional methods.

scholarly journals Kaposi sarcoma–associated herpesvirus/human herpesvirus 8–associated lymphoproliferative disorders

Blood ◽  
2019 ◽  
Vol 133 (11) ◽  
pp. 1186-1190 ◽  
Author(s):  
Eric Oksenhendler ◽  
David Boutboul ◽  
Lionel Galicier
Keyword(s):  
B Cells ◽  
Primary Effusion Lymphoma ◽  
Human Herpesvirus ◽  
Kaposi Sarcoma ◽  
Castleman Disease ◽  
Lymphoproliferative Disorders ◽  
Human Herpesvirus 8 ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Infected Cells

Abstract Kaposi sarcoma–associated herpesvirus/human herpesvirus 8 is associated with multicentric Castleman disease (MCD) and primary effusion lymphoma (PEL). In MCD, infected B cells, although polyclonal, express a monotypic immunoglobulin Mλ phenotype, probably through editing toward λ light chain in mature B cells. They are considered to originate from pre–germinal center (GC) naive B cells. Both viral and human interleukin-6 contribute to the plasmacytic differentiation of these cells, and viral replication can be observed in some infected cells. PEL cells are clonal B cells considered as GC/post-GC B cells. One can also hypothesize that they originate from the same infected naive B cells and that additional factors could be responsible for their peculiar phenotype.

High levels of human herpesvirus 8 viral load, human interleukin-6, interleukin-10, and C reactive protein correlate with exacerbation of multicentric Castleman disease in HIV-infected patients

Blood ◽  
2000 ◽  
Vol 96 (6) ◽  
pp. 2069-2073 ◽  
Author(s):  
Eric Oksenhendler ◽  
Guislaine Carcelain ◽  
Yoshiyasu Aoki ◽  
Emmanuelle Boulanger ◽  
Anne Maillard ◽  
...  
Keyword(s):  
Viral Load ◽  
Interleukin 6 ◽  
Lymphoproliferative Disorder ◽  
Human Herpesvirus ◽  
Castleman Disease ◽  
Human Herpesvirus 8 ◽  
C Reactive Protein ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Reactive Protein

Abstract Multicentric Castleman disease (MCD) is a distinct type of lymphoproliferative disorder associated with inflammatory symptoms and interleukin-6 (IL-6) dysregulation. In the context of human immunodeficiency virus (HIV) infection, MCD is associated with human herpesvirus 8 (HHV8) infection. In a prospective study of 23 HIV-infected patients with MCD, clinical symptoms of MCD were present at 45 visits, whereas patients were in chemotherapy-induced clinical remission at 50 visits. Symptoms were associated with a high level of serum C reactive protein, high HHV8 viral load in peripheral blood mononuclear cells, and high plasma human IL-6 and IL-10 levels. Strong correlations between plasma IL-6 and plasma IL-10 with the HHV8 viral load suggest that both cytokines may be involved in the pathogenesis of this virus-associated lymphoproliferative disorder.

EBV+ HHV-8+ Multicentric Castleman Disease With Plasmablastic Aggregates in an HIV+ Man: An Evolving Clinicopathologic Entity

2017 ◽  
Vol 26 (4) ◽  
pp. 338-341 ◽  
Author(s):  
Aditya Shivane ◽  
Amy Pearce ◽  
Nadia Khatib ◽  
Mark E. F. Smith
Keyword(s):  
Human Herpesvirus ◽  
Cervical Lymphadenopathy ◽  
Castleman Disease ◽  
Hiv Positive ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Barr Virus ◽  
Positive Individual ◽  
Epstein Barr

We report a case of EBV+ and HHV-8+ multicentric Castleman disease with plasmablastic aggregates in an HIV-positive individual. A 41-year-old man presented in early 2015 with fevers, sweats, weight loss, intractable itching, and on subsequent testing was found to be HIV positive. Investigations showed cervical lymphadenopathy and splenomegaly. He was treated for HIV and his symptoms resolved. His symptoms recurred in January 2016, and a provisional diagnosis of multicentric Castleman disease was entertained. The HHV-8 (human herpesvirus-8) and EBV (Epstein-Barr virus) viral load was elevated. A left supraclavicular lymph node core biopsy was performed, which showed features of multicentric Castleman disease with plasmablastic aggregates that are EBV (EBER) and HHV-8 positive. He responded well to rituximab treatment and remains well with no symptoms at recent follow-up.

High levels of human herpesvirus 8 viral load, human interleukin-6, interleukin-10, and C reactive protein correlate with exacerbation of multicentric Castleman disease in HIV-infected patients

Blood ◽  
2000 ◽  
Vol 96 (6) ◽  
pp. 2069-2073 ◽  
Author(s):  
Eric Oksenhendler ◽  
Guislaine Carcelain ◽  
Yoshiyasu Aoki ◽  
Emmanuelle Boulanger ◽  
Anne Maillard ◽  
...  
Keyword(s):  
Viral Load ◽  
Interleukin 6 ◽  
Lymphoproliferative Disorder ◽  
Human Herpesvirus ◽  
Castleman Disease ◽  
Human Herpesvirus 8 ◽  
C Reactive Protein ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Reactive Protein

Multicentric Castleman disease (MCD) is a distinct type of lymphoproliferative disorder associated with inflammatory symptoms and interleukin-6 (IL-6) dysregulation. In the context of human immunodeficiency virus (HIV) infection, MCD is associated with human herpesvirus 8 (HHV8) infection. In a prospective study of 23 HIV-infected patients with MCD, clinical symptoms of MCD were present at 45 visits, whereas patients were in chemotherapy-induced clinical remission at 50 visits. Symptoms were associated with a high level of serum C reactive protein, high HHV8 viral load in peripheral blood mononuclear cells, and high plasma human IL-6 and IL-10 levels. Strong correlations between plasma IL-6 and plasma IL-10 with the HHV8 viral load suggest that both cytokines may be involved in the pathogenesis of this virus-associated lymphoproliferative disorder.

Human herpesvirus-8: Kaposi sarcoma, multicentric Castleman disease, and primary effusion lymphoma

Hematology ◽  
2013 ◽  
Vol 2013 (1) ◽  
pp. 103-108 ◽  
Author(s):  
Lawrence D. Kaplan
Keyword(s):  
Primary Effusion Lymphoma ◽  
Human Herpesvirus ◽  
Kaposi Sarcoma ◽  
Castleman Disease ◽  
Human Herpesvirus 8 ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Cell Cycle Entry ◽  
Clinical Challenge ◽  
Gamma Herpesvirus

Abstract Human herpesvirus 8 (HHV8) is a gamma herpesvirus associated with Kaposi sarcoma, multicentric Castleman disease, and primary effusion lymphoma, lymphoproliferative diseases that are most commonly observed in immunocompromised individuals. The viral genome expresses genes responsible for inhibition of apoptosis, cell cycle entry, and angiogenesis. Viral homologs of human regulatory genes are expressed, providing stimuli for angiogenesis, B-cell proliferation, and immune evasion. Variations in expression of these factors give rise to the 3 known HHV8-associated malignancies. Identification of these pathogenetic mechanisms has led to exploration of targeted treatment approaches for all 3 of these disorders with success in Kaposi sarcoma and multicentric Castleman disease; primary effusion lymphoma remains a clinical challenge.

Plasma HHV8 DNA predicts relapse in individuals with HIV-associated multicentric Castleman disease

Blood ◽  
2011 ◽  
Vol 118 (2) ◽  
pp. 271-275 ◽  
Author(s):  
Justin Stebbing ◽  
Caroline Adams ◽  
Adam Sanitt ◽  
Salvinia Mletzko ◽  
Mark Nelson ◽  
...  
Keyword(s):  
Human Herpesvirus ◽  
Castleman Disease ◽  
Human Herpesvirus 8 ◽  
Histologic Diagnosis ◽  
Herpesvirus 8 ◽  
Multicentric Castleman Disease ◽  
Younger Age ◽  
Systemic Symptoms ◽  
Anti Cd20 ◽  
Active Attack

Abstract HIV-associated multicentric Castleman disease (HIV-MCD) is a rare lymphoproliferative disorder caused by infection with human herpesvirus-8. The disease follows a relapsing and remitting clinical course, with marked systemic symptoms during an active attack, which can prove fatal. Its incidence is rising, and new data indicate the utility of the anti-CD20 monoclonal antibody rituximab at inducing remissions in both first- and second-line settings, although biomarkers associated with relapse have not been previously identified. In 52 individuals with a histologic diagnosis of HIV-MCD, we performed univariate and multivariate analyses to predict factors associated with an HIV-MCD attack. Although a younger age (< 50 years) was associated with an attack, the strongest association was observed with plasma levels of human herpesvirus-8 DNA. Rising levels predicted an attack (hazard ratio = 2.9; 95% confidence interval, 1.3-6.7), and maintenance therapy with rituximab should be considered in these individuals.

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