Article

Brief communication: rituximab in HIV-associated multicentric Castleman disease.

Department of Oncology, Imperial College, The Chelsea and Westminster Hospital, Barts and the London NHS Trust, and Queen Mary's University, London, United Kingdom.
Annals of internal medicine (impact factor: 16.73). 01/2008; 147(12):836-9. pp.836-9
Source: PubMed

ABSTRACT HIV-associated multicentric Castleman disease is a rare lymphoproliferative disorder with marked systemic symptoms attributed to cytokine disarray. Many therapeutic approaches in small series of patients have proved largely unsuccessful to date.
To investigate the efficacy and clinicopathologic variables associated with first-line treatment for HIV-associated multicentric Castleman disease with the anti-CD20 monoclonal antibody rituximab.
Single-group, open-label, phase II trial.
3 teaching hospitals in England.
Previously untreated patients with histologically proven HIV-associated multicentric Castleman disease.
4 infusions of rituximab, 375 mg per m2 of body surface area, at weekly intervals.
Response was evaluated clinically and radiologically and by measuring plasma Kaposi sarcoma-associated herpesvirus viral load.
21 consecutive patients (18 men) with plasmablastic multicentric Castleman disease were recruited. The median follow-up was 12 months (range, 1 to 49 months). One patient died before completing therapy, 20 achieved remission of symptoms, and 14 (67%) achieved a radiologic response. The overall and disease-free survival rates at 2 years were 95% (95% CI, 86% to 100%) and 79% (CI, 49% to 100%), respectively. Plasma acute-phase proteins, immunoglobulins, and Kaposi sarcoma-associated herpesvirus viral load decreased after rituximab therapy. The main adverse effect was reactivation of Kaposi sarcoma.
The study had no comparison group.
Rituximab may be clinically valuable as initial therapy for HIV-associated multicentric Castleman disease.

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    Article: Epstein-Barr virus myelitis and Castleman's disease in a patient with acquired immune deficiency syndrome: a case report.
    Costantine Albany, George Psevdos, Jasminka Balderacchi, Victoria L Sharp
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    ABSTRACT: Few cases of Epstein-Barr virus myelitis have been described in the literature. Multi-centric Castleman's disease is a lymphoproliferative disorder that is well known for its associations with the human immunodeficiency virus, human herpes virus 8, and Kaposi's sarcoma. The concurrent presentation of these two diseases in a patient at the same time is extremely unusual. We describe the case of a 43-year-old Caucasian man with acquired immune deficiency syndrome who presented with fever, weight loss and diffuse lymphadenopathy, and was diagnosed with multi-centric Castleman's disease. He presented three weeks later with lower extremity weakness and urinary retention, at which time cerebrospinal fluid contained lymphocytic pleocytosis and elevated protein. Magnetic resonance imaging demonstrated abnormal spinal cord signal intensity over several cervical and thoracic segments, suggesting the diagnosis of myelitis. Our patient was ultimately diagnosed with Epstein-Barr virus myelitis, as Epstein-Barr virus DNA was detected by polymerase chain reaction in the cerebrospinal fluid. To the best of our knowledge, this is the first case of multi-centric Castleman's disease followed by acute Epstein-Barr virus myelitis in a human immunodeficiency virus-infected patient. Clinicians caring for human immunodeficiency virus-infected patients should be vigilant about monitoring patients with increasing lymphadenopathy, prompting thorough diagnostic investigations when necessary.
    Journal of Medical Case Reports 05/2011; 5:209.
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    Article: Pathology of rituximab-induced Kaposi sarcoma flare.
    Liron Pantanowitz, Klaus Früh, Sharon Marconi, Ashlee V Moses, Bruce J Dezube
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    ABSTRACT: Kaposi sarcoma (KS) flare may occur following therapy with corticosteroids, as part of the immune reconstitution inflammatory syndrome seen with highly active antiretroviral therapy (HAART), and after rituximab therapy. The exact mechanism responsible for iatrogenic KS flare is unclear. A case of AIDS-associated cutaneous KS flare following rituximab therapy was compared to similar controls by means of immunohistochemistry using vascular makers (CD34, CD31), monoclonal antibodies to Human Herpesvirus 8 (HHV8) gene products (LNA-1, K5), as well as B-lymphocyte (CD20) and T-lymphocyte (CD3, CD4, CD8) markers. CD20+ B-cell depletion with rituximab in KS flare occurred concomitantly with activation of the HHV8 immediate early gene protein K5. KS flare in this patient was successfully treated with liposomal doxorubicin and valganciclovir. Rituximab-induced KS flare appears to be related to HHV8 activation. Effective management of iatrogenic KS flare therefore depends upon the control of HHV8 viremia in conjunction with specific chemotherapy for KS.
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Keywords

21 consecutive patients
 
4 infusions
 
49 months
 
anti-CD20 monoclonal antibody rituximab
 
body surface area
 
clinicopathologic variables
 
disease-free survival rates
 
HIV-associated multicentric Castleman disease
 
initial therapy
 
main adverse effect
 
median follow-up
 
phase II trial
 
Plasma acute-phase proteins
 
plasmablastic multicentric Castleman disease
 
radiologic response
 
rituximab therapy
 
systemic symptoms
 
therapeutic approaches
 
untreated patients
 
weekly intervals