Prospective Study of Rituximab in Chemotherapy-Dependent Human Immunodeficiency Virus–Associated Multicentric Castleman's Disease: ANRS 117 CastlemaB Trial

Gérard, Laurence; Bérezné, Alice; Galicier, Lionel; Meignin, Véronique; Obadia, Martine; Castro, Nathalie De; Jacomet, Christine; Verdon, Renaud; Madelaine-Chambrin, Isabelle; Boulanger, Emmanuelle; Chevret, Sylvie; Agbalika, F.; Oksenhendler, Éric

doi:10.1200/jco.2007.10.6732

Cited by 207 publications

(195 citation statements)

References 26 publications

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“…A similar difference was also observed by Table 2 Comparative summarisation of prominent large case series published in the recent past Mylona et al [1] Gerard et al [6] Bower et al [10] Luo et al [8] Luo et al [8], who noted median age for UCD at 30 years and for MCD at 41 years. A strong predilection of CD for male patients was reported in earlier studies with M:F ratio as high as 9:1 [1,6,17]. We also found M:F ratio to be in favour of males (2.2:1).…”

Section: Discussionsupporting

confidence: 72%

“…The fact that many of the cases reviewed by Mylona et al were treated in the pre-ART era may have contributed to the poorer survival of HIV-related CD. In the current era with the advent of modern ART survival rates of MCD in HIV-positive patients have considerably improved, and they are nearing survival reported in immunocompetent patients [6,8,10,13] (Table 2).…”

Section: Discussionmentioning

confidence: 97%

“…A wide range of options ranging from observation, wide local excision, single-agent chemotherapy to multi-agent chemotherapy with or without stem cell transplants are described in the literature [1,6,8,10] with relatively satisfactory outcome (Table 2). In our study, there was a significant difference in treatment response amongst UCD and MCD.…”

Section: Discussionmentioning

confidence: 99%

“…Over the last few decades, gradual rise in the incidence of multicentric CD is noted, and this trend is parallel to the rise in HIV infections along with increasing use of antiretroviral therapy [2]. The majority of the published studies on CD discuss presentation and outcome of CD in patients suffering from HIV [1,2,6]. The data on presentation and treatment outcome in patients with CD in immunocompetent hosts is very limited.…”

Section: Introductionmentioning

confidence: 99%

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Presentation and Outcome of Castleman’s Disease in Immunocompetent Hosts

Prakash¹,

Bal

Malhotra³

et al. 2015

Indian J Hematol Blood Transfus

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Castleman's disease (CD) is a heterogeneous lymphoproliferative disorder of unknown aetiology. Mostly, this disorder is seen in immunocompromised hosts. It is known to be associated with systemic disorders like HIV, HHV-8, lymphoma, and Kaposi sarcoma. As of today, the clinical behaviour and outcome of CD in immunocompetent host remains suboptimally studied. We analyzed consecutively treated cases of CD presented to our centre in last 12 years. Case record files were studied for patient's characteristics, clinical presentation, baseline laboratory and pathologic parameters, therapy and outcome. This study describes presentation and treatment outcome of CD in immunocompetent patients. Total 16 patients of CD were treated during the study period. The median age of patients at the time of presentation was 40.5 years (range 13-72 years). An equal number of patients (8 each) had unicentric and multicentric CD. Sixty-three percent patients had hyaline vascular subtype while 37 % patients had plasma cell or mixed variant.Majority of the patients had good performance status (ECOG PS 0, 1 in 10 (62.5 %) patients; PS2-4 in 6 (37.5 %) patients). The median duration of symptoms was 6 months (range 2-36 months). None of the patients in our study had associated HIV infection. Six patients presented with fever, out of which four had plasma cell variant of CD and three of them had multicentric involvement. In comparison to unicentric CD, patients with multicentric CD had lower albumin levels (4.15 vs. 3.38 g/dl, p = 0.006), haemoglobin levels (11.3 vs. 9.8, p = 0.06), and lower complete remission rates (62.5 % vs. none). Patients were treated according to the stage and clinical status with surgery, chemotherapy or combination of both modalities. Surgery was the predominant treatment for unicentric CD while multicentric CD was treated with various chemotherapy regimens. Eight patients were treated with chemotherapy (CHOP-based regimen-5, melphalan-prednisolone thalidomide-1, chlorambucil-prednisolone-1, and only corticosteroids-1). In the entire study group, ORR was 72 % (CR 36 %, PR 36 %), one patient died of progressive disease during chemotherapy. Two patients lost to followup before assessment of treatment response. Five-year overall survival was 100 and 87 % for unicentric and multicentric CD respectively. Castleman's disease is a rare lymphoproliferative disorder amongst Indian patients. It more commonly presents as hyaline vascular variant and affects middle-aged individuals. The most remarkable fact in our analysis was a lack of HIV positivity in any of the patient that is in contrast to the majority of the published literature. With multimodality therapy, high response rates and long-term survival were noted in the entire study group.

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Section: Discussionsupporting

confidence: 72%

Section: Discussionmentioning

confidence: 97%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Presentation and Outcome of Castleman’s Disease in Immunocompetent Hosts

Prakash¹,

Bal

Malhotra³

et al. 2015

Indian J Hematol Blood Transfus

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“…In MCD, HHV-8 infects plasmablasts located in the mantle zone, which variably express the CD20 surface antigen (19). This has provided the rationale for using rituximab, which has shown promising responses in two prospective trials in MCD in the HIV setting (20,21). While the effect of rituximab on HHV-8 viremia has not been reported, in patients with PTLD rituximab has been shown to reduce EBV viremia, which infects the same plasmablasts as HHV-8.…”

Section: Discussionmentioning

confidence: 99%

Successful Treatment of Iatrogenic Multicentric Castleman’s Disease Arising Due to Recrudescence of HHV-8 in a Liver Transplant Patient

Speicher

Sehu

Mollee

et al. 2014

American Journal of Transplantation

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We describe the case of a 59‐year‐old HIV‐negative male who developed multicentric Castleman's disease (MCD) 1 year postliver transplantation due to recrudescence of a pretransplant human herpesvirus‐8 (HHV‐8) infection. He presented with fevers, dry cough, weight loss and drenching night sweats. Routine investigations were all unremarkable. Computerized axial tomography (CT) scans showed splenomegaly and intra‐abdominal lymphadenopathy, confirmed by positron emission tomography. Cervical lymph node biopsies were consistent with MCD. The presence of HHV‐8 was confirmed on immunohistochemistry. Peripheral blood HHV‐8 quantitative polymerase chain reaction (qPCR) monitoring showed a threefold decrease in viremia in the first week of treatment with ganciclovir but had little impact on clinical symptoms. Reducing immunosuppression and switching to rituximab resolved clinical symptoms and produced a negative HHV‐8 qPCR result. Retrospective molecular testing of sera collected pre‐ and immediately posttransplantation confirmed preexisting HHV‐8 in the host. This is the first reported case of an HIV‐negative postliver transplant patient developing MCD that manifested as posttransplant lymphoproliferative disorder due to recrudescence of HHV‐8. We propose (1) the introduction of the term iatrogenic Castleman's disease (CD) for this and similar cases, (2) rituximab should be considered as a treatment option for CD and (3) consideration be given to a change to the World Health Organization classification of CD to incorporate such cases.

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