Correspondence
An Unusual Cluster of Cases of Castleman's Disease during Highly Active Antiretroviral Therapy for AIDS
N Engl J Med 1999; 340:1923-1924June 17, 1999
- Article
To the Editor:
Castleman and coworkers first described a lymphatic hyperplasia now called Castleman's disease, which has two forms, the hyaline vascular type (found in 90 percent of cases) and the plasma-cell type (found in 10 percent).1,2 Patients may present with a multicentric lymphadenopathy and progressive systemic symptoms or with a more localized, indolent disease that can often be cured by local excision. The majority of patients with so-called multicentric Castleman's disease have the plasma-cell–rich variant with an interfollicular predominance of plasma cells on histologic analysis.2,3 Multicentric Castleman's disease is associated with infection with human herpesvirus 8 (HHV-8)4,5 and is occasionally found with Kaposi's sarcoma.3-5
Recently, we observed an unusual cluster of cases of rapidly progressing multicentric Castleman's disease. Fever, weakness, generalized enlargement of lymph nodes, and marked polyclonal gammopathy developed in three patients with AIDS. Lymph-node biopsies established the diagnosis of plasma-cell–rich multicentric Castleman's disease (Figure 1AFigure 1
A Lymph-Node–Biopsy Specimen from a Patient with Multicentric Castleman's Disease. and Figure 1B). All patients proved to be positive for HHV-8; two of the patients had a history of Kaposi's sarcoma, and the third had sarcomatoid Kaposi's sarcoma lesions along with evidence of multicentric Castleman's disease in the same lymph nodes. Two of these patients died within one week after the diagnosis, with generalized involvement of the lymphatic system, liver, and bone marrow at autopsy. A fourth patient with AIDS who died equally rapidly after the diagnosis of multicentric Castleman's disease had been seen in our hospital 14 months earlier.This cluster of cases of rapidly progressive multicentric Castleman's disease is unusual and important for three reasons. First, although several cases of this disease have been reported in patients with AIDS, it remains a rare disease, especially in areas with a low seroprevalence of HHV-8. It is unknown whether this cluster is a chance observation or a sign of an increasing incidence of multicentric Castleman's disease, possibly associated with changes in the virulence and cellular tropism of HHV-8 and human immunodeficiency virus type 1. Second, these case reports show that multicentric Castleman's disease, especially in patients with AIDS, should be handled as a medical emergency. Third, symptoms of multicentric Castleman's disease started after the initiation of highly active antiretroviral therapy in these three patients. For safety's sake, all physicians prescribing highly active antiretroviral therapy should monitor their HHV-8–positive patients for signs and symptoms of this disease.
5 ReferencesChristian Zietz, M.D.
Johannes R. Bogner, M.D.
Frank-Detlef Goebel, M.D.
Udo Löhrs, M.D.
Ludwig Maximilians University, 80337 Munich, Germany1
Castleman B ,Iverson L ,Menendez VP . Localized mediastinal lymph-node hyperplasia resembling thymoma. Cancer 1956;9:822-830
CrossRef | Web of Science | Medline2
Keller AR ,Hochholzer L ,Castleman B . Hyaline-vascular and plasma-cell types of giant lymph node hyperplasia of the mediastinum and other locations. Cancer 1972;29:670-683
CrossRef | Web of Science | Medline3
Peterson BA ,Frizzera G . Multicentric Castleman's disease. Semin Oncol 1993;20:636-647
Web of Science | Medline4
Dupin N ,Gorin I ,Deleuze J ,Agut H ,Huraux J-M ,Escande J-P . Herpes-like DNA sequences, AIDS-related tumors, and Castleman's disease. N Engl J Med 1995;333:798-798
Web of Science | Medline5
Soulier J ,Grollet L ,Oksenhendler E , et al. Kaposi's sarcoma-associated herpesvirus-like DNA sequences in multicentric Castleman's disease. Blood 1995;86:1276-1280
Web of Science | Medline
- Citing Articles (31)
Citing Articles
1
C. Hoffmann, H. Schmid, M. Muller, C. Teutsch, J. van Lunzen, S. Esser, T. Wolf, C. Wyen, M. Sabranski, H.-A. Horst, S. Reuter, M. Vogel, H. Jager, J. Bogner, K. Arasteh. (2011) Improved outcome with rituximab in patients with HIV-associated multicentric Castleman disease. Blood 118:13, 3499-3503
CrossRef2
Deepa Reddy, Ronald Mitsuyasu. (2011) HIV-associated multicentric Castleman disease. Current Opinion in Oncology 23:5, 475-481
CrossRef3
M. Bower. (2010) How I treat HIV-associated multicentric Castleman disease. Blood 116:22, 4415-4421
CrossRef4
Klaus-Martin Schulte, Nadia Talat. (2010) Castleman's disease—a two compartment model of HHV8 infection. Nature Reviews Clinical Oncology 7:9, 533-543
CrossRef5
Bernhard Fleckenstein, Frank Neipel. 2010. Human Herpesvirus 8. .
CrossRef6
C. Simonelli, R. Tedeschi, A. Gloghini, R. Talamini, M.T. Bortolin, M. Berretta, M. Spina, S. Morassut, E. Vaccher, P. De Paoli, A. Carbone, U. Tirelli. (2009) Plasma HHV-8 viral load in HHV-8-related lymphoproliferative disorders associated with HIV infection. Journal of Medical Virology 81:5, 888-896
CrossRef7
Eric Oksenhendler. (2009) HIV-associated multicentric Castleman disease. Current Opinion in HIV and AIDS 4:1, 16-21
CrossRef8
Ren-rong Tian, Qing-jiao Liao, Xulin Chen. (2008) Prevention and treatment of KSHV-associated diseases with antiviral drugs. Virologica Sinica 23:6, 486-495
CrossRef9
Justin Stebbing, Liron Pantanowitz, Farshid Dayyani, Ryan J. Sullivan, Mark Bower, Bruce J. Dezube. (2008) HIV-associated multicentric Castleman's disease. American Journal of Hematology 83:6, 498-503
CrossRef10
Bouvresse Sophie, Marcelin Anne-Genevi??ve, Franck Nathalie, Regnier St??phanie, Damond Florence, Tulliez Micheline, Dupin Nicolas. (2007) The first reported case and management of multicentric Castleman??s disease associated with Kaposi??s sarcoma in an HIV-2-infected patient. AIDS 21:11, 1492-1494
CrossRef11
Anu Dham, Bruce A Peterson. (2007) Castleman disease. Current Opinion in Hematology 14:4, 354-359
CrossRef12
Makoto Ide, Yasunori Kawachi, Yoichiro Izumi, Kanji Kasagi, Tetsuro Ogino. (2006) Long-term remission in HIV-negative patients with multicentric Castleman's disease using rituximab. European Journal of Haematology 76:2, 119-123
CrossRef13
Rom S. Leidner, David M. Aboulafia. (2005) Recrudescent Kaposi's Sarcoma After Initiation of HAART: A Manifestation of Immune Reconstitution Syndrome. AIDS Patient Care and STDs 19:10, 635-644
CrossRef14
Rom S. Leidner, David M. Aboulafia. (2005) Recrudescent Kaposi's Sarcoma After Initiation of HAART: A Manifestation of Immune Reconstitution Syndrome. AIDS Patient Care and STDs 19:10, 849-858
CrossRef15
M. Eulalia Valencia, Victoria Moreno, Pilar Martínez, Isabel Casado. (2005) Evolución favorable de la enfermedad de Castleman tratada con valganciclovir oral. Medicina Clínica 125:10, 399
CrossRef16
Corey Casper. (2005) The aetiology and management of Castleman disease at 50 years: translating pathophysiology to patient care. British Journal of Haematology 129:1, 3-17
CrossRef17
Matthias Stoll, Reinhold E. Schmidt. (2004) Immune restoration inflammatory syndromes: Apparently paradoxical clinical events after the initiation of HAART. Current HIV/AIDS Reports 1:3, 122-127
CrossRef18
Martyn A French, Patricia Price, Shelley F Stone. (2004) Immune restoration disease after antiretroviral therapy. AIDS 18:12, 1615-1627
CrossRef19
Matthias Stoll, Reinhold E Schmidt. (2004) Adverse events of desirable gain in immunocompetence: the Immune Restoration Inflammatory Syndromes. Autoimmunity Reviews 3:4, 243-249
CrossRef20
Ilija Tomic, Goran Plavec, Olga Tasic, Aleksandar Ristanovic, Vlado Cvijanovic. (2004) Transitional type of Castleman's disease manifested as the POEMS syndrome. Vojnosanitetski pregled 61:4, 439-444
CrossRef21
Andrew Godfrey, Heike Laman, Chris Boshoff. (2003) RNA interference: a potential tool against Kaposi??s sarcoma-associated herpesvirus. Current Opinion in Infectious Diseases 16:6, 593-600
CrossRef22
Matthias Stoll, Reinhold E. Schmidt. (2003) Immune restoration inflammatory syndromes: The dark side of successful antiretroviral treatment. Current Infectious Disease Reports 5:3, 266-276
CrossRef23
Yoshiyasu Aoki, Giovanna Tosato. (2003) Pathogenesis and manifestations of human herpesvirus-8-associated disorders. Seminars in Hematology 40:2, 143-153
CrossRef24
Ulrich R Hengge, Thomas Ruzicka, Stephen K Tyring, Martin Stuschke, Michael Roggendorf, Robert A Schwartz, Siegfried Seeber. (2002) Update on Kaposi's sarcoma and other HHV8 associated diseases. Part 2: pathogenesis, Castleman's disease, and pleural effusion lymphoma. The Lancet Infectious Diseases 2:6, 344-352
CrossRef25
Tadeusz Robak. (2002) The Role of Nucleoside Analogues in the Treatment of Chronic Lymphocytic Leukemia-Lessons Learned from Prospective Randomized Trials. Leukemia & Lymphoma 43:3, 537-548
CrossRef26
SF Stone, P Price, NM Keane, RJ Murray, MA French. (2002) Levels of IL-6 and soluble IL-6 receptor are increased in HIV patients with a history of immune restoration disease after HAART. HIV Medicine 3:1, 21-27
CrossRef27
Elizabeth Connick. (2001) Immune Reconstitution in HIV-1-Infected Individuals Treated with Potent Antiretroviral Therapy. Journal of Investigative Dermatology Symposium Proceedings 6:3, 212-218
CrossRef28
T. Robak, H. Urbanska-Rys, B. Strzelecka, E. Krykowski, J. Bartkowiak, J. Z. Bonski, R. Kordek, K. Warzocha. (2001) Plasmablastic lymphoma in a patient with chronic lymphocytic leukemia heavily pretreated with cladribine (2-CdA): an unusual variant of Richter's syndrome. European Journal of Haematology 67:5-6, 322-327
CrossRef29
Valeria Ascoli, Sabina Signoretti, Andrea Onetti-Muda, Edoardo Pescarmona, Carlo Della-Rocca, Francesco Nardi, Claudio Maria Mastroianni, Roberta Gastaldi, Alessandra Pistilli, Gianluca Gaidano, Antonino Carbone, Francesco Lo-Coco. (2001) Primary effusion lymphoma in HIV-infected patients with multicentric Castleman's disease. The Journal of Pathology 193:2, 200-209
CrossRef30
Carlos Zala, Clawdia Ochoa, Alejandro Krolewiecki, Patricia Patterson, Pedro Cahn, Richard I. Crawford, Julio S. G. Montaner. (2000) Highly active antiretroviral therapy does not protect against Kaposi's sarcoma in HIV-infected individuals. AIDS 14:14, 2217
CrossRef31
P. Kumari, G. P. Schechter, N. Saini, D. A. Benator. (2000) Successful Treatment of Human Immunodeficiency Virus-Related Castleman's Disease with Interferon- . Clinical Infectious Diseases 31:2, 602-604
CrossRef
