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• W2107971057 abstract "The use of highly active antiretroviral therapy (HAART) was associated with a dramatic improvement in the outcome of patients with human immunodeficiency virus (HIV) infection [1.Palella Jr., F.J. Delaney K.M. Moorman A.C. et al.Declining morbidity and mortality among patients with advanced human immunodeficiency virus infection.N Engl J Med. 1998; 338: 853-860Crossref PubMed Scopus (8475) Google Scholar, 2.Miller V. Staszewski S. Nisius G. et al.Risk of new AIDS diseases in people on triple therapy.Lancet. 1999; 353: 463-464Abstract Full Text Full Text PDF PubMed Scopus (40) Google Scholar]. In this report, we would like to describe the case of a patient with AIDS-related multicentric Castleman’s disease who has also enjoyed a long-lasting complete clinical and pathological remission following the use of HAART alone.Castleman’s disease belongs to the group of atypical lymphoid proliferations, which are usually confused with the diagnosis of malignant lymphoma. First described in 1956, the typical patient presents with either localized mediastinal lymphadenopathy or a more aggressive form of the disease characterized by diffuse lymphadenopathy and systemic symptoms [3.Castleman B. Iverson L. Menendez V.P. Localized mediastinal lymph-node hyperplasia resembling thymoma.Cancer. 1956; 9: 822-830Crossref PubMed Scopus (1252) Google Scholar]. The differential diagnosis of Castleman’s disease should include malignant lymphoma, as well as other causes of lymphoma-like syndromes, such as adverse reactions to drugs, autoimmune disorders and viral or bacterial infections.Until recently, Castleman’s disease was not considered an AIDS-related event, being managed with local surgery, irradiation and cortisteroids. Cytotoxic therapy was reserved only for highly symptomatic patients with refractory disease. Over the past years, however, a newly described AIDS-related multicentric form of Castleman’s disease (MCD) has been recognized. In this aggressive form of the disease, patients develop progressive lymphadenopathy, B symptoms and usually a fatal course [4.Oksenhendler E. Duarte M. Soulier J. et al.Multicentric Castleman’s disease in HIV infection: a clinical and pathological study of 20 patients.AIDS. 1996; 10: 61-67Crossref PubMed Scopus (326) Google Scholar].Our patient was a 46 year-old HIV-positive black man who presented with dyspnea, cough, hemoptysis and severe weight loss. He had fever, bibasilar rales and widespread lymphadenopathy. The blood tests were normal, except for a hemoglobin level of 10.7 g/dl. The chest X-ray showed bilateral reticulonodular infiltrates and the computed tomography (CT) scan revealed multiple mediastinal lymph nodes (Figure 1a). The CD4 count was 54 cells/mm3 and the CD4/CD8 ratio was 0.06. A supraclavicular and submandibular lymph node biopsy confirmed MCD. The additional medical work-up ruled out malignant lymphoma, catch-scratch fever, autoimmune or infectious diseases.Due to the aggressiveness of the disease, combination chemotherapy was offered to the patient, which he refused due to fear of infectious complications. HAART (lamivudine/zidovudine plus indinavir/ritonavir) alone was started and, surprisingly, the patient showed a rapid clinical improvement, with the disappearance of B symptoms within days. By the end of an 18-month follow-up period, the CT scan showed a complete disappearance of the mediastinal lymph nodes (Figure 1b) and a new lymph node biopsy revealed only reactive changes. The CD4 count was135 cells/mm3 and the viral load was less than 50 copies/ml. The patient remains completely asymptomatic with no evidence of Castleman’s disease following a 24 month follow-up period on HAART alone.Our report comes as the first in which a complete histologically-proven response for a prolonged period of follow-up is clearly documented in AIDS-related MCD with the use of HAART alone. There are other reports on the clinical outcome of AIDS-related MCD, using a variety of combined therapeutic approaches including antiviral drugs, interferon-α, anti-interleukin-6 and chemotherapeutic agents along with HAART. In general, tumor responses are partial and short-lasting, sometimes at the cost of significant clinical toxicity [5.Lanzafame M. Carretta G. Trevenzoli M. et al.Successful treatment of Castleman’s disease with HAART in two HIV-infected patients.J Infect Dis. 2000; 40: 90-91Scopus (26) Google Scholar, 6.Revuelta M.P. Nord J.A. Successful treatment of multicentric Castleman’s disease in a patient with human immunodeficiency virus.Clin Infect Dis. 1998; 26: 527Crossref PubMed Scopus (15) Google Scholar, 7.Scott D. Cabral L. Harrington Jr, W.J. Treatment of HIV-associated multicentric Castleman’s disease with oral etoposide.Am J Hematol. 2001; 66: 148-150Crossref PubMed Scopus (65) Google Scholar, 8.Dupin N. Krivine A. Calvez V. et al.No effect of protease inhibitor on clinical and virological evolution of Castleman’s disease in an HIV-1 infected patient.AIDS. 1997; 11: 1400-1401Google Scholar]. It should be noted that our patient did not develop a clear neoplastic transformation of the disease into a malignant lymphoma after such a long-term follow-up period. Considering the poor treatment results and high risk of infectious complications following cytotoxic therapy in the AIDS population, we postulate that patients with AIDS-related MCD should be first managed with HAART, leaving more aggressive therapeutic approaches for patients at relapse. The use of highly active antiretroviral therapy (HAART) was associated with a dramatic improvement in the outcome of patients with human immunodeficiency virus (HIV) infection [1.Palella Jr., F.J. Delaney K.M. Moorman A.C. et al.Declining morbidity and mortality among patients with advanced human immunodeficiency virus infection.N Engl J Med. 1998; 338: 853-860Crossref PubMed Scopus (8475) Google Scholar, 2.Miller V. Staszewski S. Nisius G. et al.Risk of new AIDS diseases in people on triple therapy.Lancet. 1999; 353: 463-464Abstract Full Text Full Text PDF PubMed Scopus (40) Google Scholar]. In this report, we would like to describe the case of a patient with AIDS-related multicentric Castleman’s disease who has also enjoyed a long-lasting complete clinical and pathological remission following the use of HAART alone. Castleman’s disease belongs to the group of atypical lymphoid proliferations, which are usually confused with the diagnosis of malignant lymphoma. First described in 1956, the typical patient presents with either localized mediastinal lymphadenopathy or a more aggressive form of the disease characterized by diffuse lymphadenopathy and systemic symptoms [3.Castleman B. Iverson L. Menendez V.P. Localized mediastinal lymph-node hyperplasia resembling thymoma.Cancer. 1956; 9: 822-830Crossref PubMed Scopus (1252) Google Scholar]. The differential diagnosis of Castleman’s disease should include malignant lymphoma, as well as other causes of lymphoma-like syndromes, such as adverse reactions to drugs, autoimmune disorders and viral or bacterial infections. Until recently, Castleman’s disease was not considered an AIDS-related event, being managed with local surgery, irradiation and cortisteroids. Cytotoxic therapy was reserved only for highly symptomatic patients with refractory disease. Over the past years, however, a newly described AIDS-related multicentric form of Castleman’s disease (MCD) has been recognized. In this aggressive form of the disease, patients develop progressive lymphadenopathy, B symptoms and usually a fatal course [4.Oksenhendler E. Duarte M. Soulier J. et al.Multicentric Castleman’s disease in HIV infection: a clinical and pathological study of 20 patients.AIDS. 1996; 10: 61-67Crossref PubMed Scopus (326) Google Scholar]. Our patient was a 46 year-old HIV-positive black man who presented with dyspnea, cough, hemoptysis and severe weight loss. He had fever, bibasilar rales and widespread lymphadenopathy. The blood tests were normal, except for a hemoglobin level of 10.7 g/dl. The chest X-ray showed bilateral reticulonodular infiltrates and the computed tomography (CT) scan revealed multiple mediastinal lymph nodes (Figure 1a). The CD4 count was 54 cells/mm3 and the CD4/CD8 ratio was 0.06. A supraclavicular and submandibular lymph node biopsy confirmed MCD. The additional medical work-up ruled out malignant lymphoma, catch-scratch fever, autoimmune or infectious diseases. Due to the aggressiveness of the disease, combination chemotherapy was offered to the patient, which he refused due to fear of infectious complications. HAART (lamivudine/zidovudine plus indinavir/ritonavir) alone was started and, surprisingly, the patient showed a rapid clinical improvement, with the disappearance of B symptoms within days. By the end of an 18-month follow-up period, the CT scan showed a complete disappearance of the mediastinal lymph nodes (Figure 1b) and a new lymph node biopsy revealed only reactive changes. The CD4 count was135 cells/mm3 and the viral load was less than 50 copies/ml. The patient remains completely asymptomatic with no evidence of Castleman’s disease following a 24 month follow-up period on HAART alone. Our report comes as the first in which a complete histologically-proven response for a prolonged period of follow-up is clearly documented in AIDS-related MCD with the use of HAART alone. There are other reports on the clinical outcome of AIDS-related MCD, using a variety of combined therapeutic approaches including antiviral drugs, interferon-α, anti-interleukin-6 and chemotherapeutic agents along with HAART. In general, tumor responses are partial and short-lasting, sometimes at the cost of significant clinical toxicity [5.Lanzafame M. Carretta G. Trevenzoli M. et al.Successful treatment of Castleman’s disease with HAART in two HIV-infected patients.J Infect Dis. 2000; 40: 90-91Scopus (26) Google Scholar, 6.Revuelta M.P. Nord J.A. Successful treatment of multicentric Castleman’s disease in a patient with human immunodeficiency virus.Clin Infect Dis. 1998; 26: 527Crossref PubMed Scopus (15) Google Scholar, 7.Scott D. Cabral L. Harrington Jr, W.J. Treatment of HIV-associated multicentric Castleman’s disease with oral etoposide.Am J Hematol. 2001; 66: 148-150Crossref PubMed Scopus (65) Google Scholar, 8.Dupin N. Krivine A. Calvez V. et al.No effect of protease inhibitor on clinical and virological evolution of Castleman’s disease in an HIV-1 infected patient.AIDS. 1997; 11: 1400-1401Google Scholar]. It should be noted that our patient did not develop a clear neoplastic transformation of the disease into a malignant lymphoma after such a long-term follow-up period. Considering the poor treatment results and high risk of infectious complications following cytotoxic therapy in the AIDS population, we postulate that patients with AIDS-related MCD should be first managed with HAART, leaving more aggressive therapeutic approaches for patients at relapse." @default.
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• W2107971057 title "Successful treatment of AIDS-related Castleman’s disease following the administration of highly active antiretroviral therapy (HAART)" @default.
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