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      Reversible rituximab-induced rectal Kaposi’s sarcoma misdiagnosed as ulcerative colitis in a patient with HIV-negative follicular lymphoma

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          Abstract

          Background

          Kaposi’s sarcoma is a low-grade mesenchymal angioproliferative tumor, most commonly observed in immunocompromised individuals, such as HIV-infected patients. Iatrogenic Kaposi’s sarcoma occurs in patients undergoing immunosuppressive therapies. Rituximab is a chimeric monoclonal antibody targeted against the pan B cell marker CD20. Because of its immunosuppressive effects through reduction of mature B-cells, it may exacerbate Kaposi’s sarcoma in HIV-positive patients. Rituximab-related Kaposi’s sarcomas have been previously reported in only two HIV-negative patients and were treated surgically.

          Case presentation

          Here, we report on a Kaposi’s sarcoma that developed under rituximab treatment in a HIV-negative 55-year-old patient treated for follicular lymphoma. The lesion developed during the maintenance rituximab therapy at the rectal level with an aspect of apparent ulcerative colitis, without any cutaneous lesion. The premature stop of rituximab led to the complete regression of Kaposi’s sarcoma, without any additional specific treatment.

          Conclusions

          To our knowledge, this is the third case of Kaposi’s sarcoma diagnosed under rituximab in a HIV-negative patient, the first one at the rectal level and the first one that completely regresses after stop of rituximab. This case raises awareness of iatrogenic Kaposi’s sarcoma in HIV-negative patients treated with rituximab, and further highlights the importance of immunosuppression in the pathophysiology of disease.

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          Most cited references27

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          Kaposi's sarcoma.

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            New treatment options have changed the survival of patients with follicular lymphoma.

            The natural history of follicular lymphoma is believed not to have changed over the last 30 years. Median survivals have ranged from 7 to 10 years, and the disease is considered incurable. However, multiple new treatment options have been developed in the last decade, and their impact on survival of follicular lymphoma remains unknown. In the current analysis, we identified all previously untreated, advanced-stage, follicular lymphoma patients treated with the following three sequential treatment approaches: cyclophosphamide, doxorubicin, vincristine, and prednisone (CHOP) chemotherapy +/- nonspecific immunostimulants (Southwest Oncology Group [SWOG] 7426 and 7713: 1974 to 1983); prednisone, methotrexate, doxorubicin, cyclophosphamide, and etoposide (ProMACE) plus mechlorethamine, vincristine, procarbazine, and prednisone (MOPP) +/- interferon (SWOG 8809: 1988 to 1994); and CHOP followed by monoclonal antibody (MoAb) therapy (SWOG 9800 and 9911: 1998 to 2000). We assessed the patients' progression-free survival (PFS) and overall survival (OS). The MoAb trials included CHOP followed by rituximab (SWOG 9800) and CHOP followed by (131)I-tositumomab (SWOG 9911). The PFS curves for the CHOP and ProMACE-MOPP studies are overlapping, with 4-year PFS estimates of 46% and 48%, respectively. However, the PFS rate of the CHOP + MoAb studies is significantly improved at 61% (P = .005). The OS curves show improvement with each succeeding study. The 4-year estimate of OS is 69% for the CHOP regimens, 79% for the ProMACE-MOPP study, and 91% for the CHOP + MoAb regimens (P < .001). These conclusions were retained after adjusting for differences in prognostic factors between the study groups. The results of this study suggest that OS for patients with follicular lymphoma has improved over time and that the choice of initial therapy may matter.
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              Rituximab decreases the risk of lymphoma in patients with HIV-associated multicentric Castleman disease.

              HIV-associated multicentric Castleman disease (MCD) is associated with a high risk of developing non-Hodgkin lymphoma (NHL). Rituximab is effective in HIV-MCD, but its impact on NHL incidence remains unknown. From a single-center prospective cohort, 113 patients were identified with a diagnosis of HIV-MCD for the present study. To compare the incidence of NHL between patients who had received a rituximab-based treatment (R+ group) and those who had not (R- group), data were analyzed before and after matching on propensity scores and after multiple imputation. The mean follow-up was 4.2 years. In the R- group (n = 65), 17 patients developed NHL (incidence, 69.6 of 1000 person years). In the R+ group (n = 48), only 1 patient developed NHL (incidence, 4.2 of 1000 person years). Based on the propensity score-matching method, a significant decrease in the incidence of NHL was observed in patients who had been treated with rituximab (hazard ratio, 0.09; 95% confidence interval, 0.01-0.70). Ten Kaposi sarcoma (KS) exacerbations and 1 newly diagnosed KS were observed in 9 patients after rituximab therapy. Rituximab was associated with an 11-fold lower risk of developing lymphoma. KS exacerbation was the most challenging adverse event after rituximab therapy.
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                Author and article information

                Contributors
                emilien.billon@free.fr
                stoppaam@ipc.unicancer.fr
                mescaml@ipc.unicancer.fr
                mbocci@ch-aubagne.fr
                monneura@ipc.unicancer.fr
                louveld@ipc.unicancer.fr
                33 4 91 22 35 37 , bertuccif@ipc.unicancer.fr
                Journal
                Clin Sarcoma Res
                Clin Sarcoma Res
                Clinical Sarcoma Research
                BioMed Central (London )
                2045-3329
                11 June 2018
                11 June 2018
                2018
                : 8
                : 11
                Affiliations
                [1 ]ISNI 0000 0004 0598 4440, GRID grid.418443.e, INSERM UMR1068, CNRS UMR725, Department of Medical Oncology, Centre de Recherche en Cancérologie de Marseille (CRCM), , Institut Paoli-Calmettes, ; 232 Bd de Sainte-Marguerite, 13009 Marseille, France
                [2 ]ISNI 0000 0004 0598 4440, GRID grid.418443.e, Department of Hematology, , Institut Paoli-Calmettes, ; Marseille, France
                [3 ]ISNI 0000 0004 0598 4440, GRID grid.418443.e, Department of Pathology, , Institut Paoli-Calmettes, ; Marseille, France
                [4 ]ISNI 0000 0001 0069 9008, GRID grid.414047.2, Department of Digestive Endoscopy Centre Hospitalier Edmond Garcin, ; Aubagne, France
                [5 ]ISNI 0000 0001 2176 4817, GRID grid.5399.6, Faculty of Medicine, , Aix-Marseille University, ; Marseille, France
                [6 ]French Sarcoma Group, Paris, France
                Article
                97
                10.1186/s13569-018-0097-7
                5994652
                29992013
                04e9eebe-9700-4e93-b58a-ed47b27da23a
                © The Author(s) 2018

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 10 November 2017
                : 13 March 2018
                Categories
                Research
                Custom metadata
                © The Author(s) 2018

                Oncology & Radiotherapy
                follicular lymphoma,immune suppression,kaposi sarcoma,rituximab
                Oncology & Radiotherapy
                follicular lymphoma, immune suppression, kaposi sarcoma, rituximab

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