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      Endobronchial Epstein-Barr Virus Associated Post-transplant Lymphoproliferative Disorder in Hematopoietic Stem Cell Transplantation

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          Abstract

          The Epstein-Barr virus (EBV) associated Post-Transplant Lymphoproliferative Disorders (PTLD) are increasingly recognized as a fatal complication of hematological stem cell transplantation (HSCT). Thoracic involvement, that may be isolated or part of a disseminated disease, usually encompasses pulmonary nodules or masses and mediastinal lymph node enlargement.

          The current case study presents 2 patients who underwent HSCT, one allogenic and the other autologous, who developed an exceptional endobronchial EBV related PTLD. The first patient had a fleshy white endobronchial mass resulting in a right upper lobe atelectasis and the second had an extensive necrotising mucosa from trachea to both basal bronchi without any significant change of lung parenchyma on the CT scan. In both cases, the diagnosis was made by bronchial biopsies. Physicians should be aware of an endobronchial pattern of EBV associated PTLD after HSCT to permit quick diagnosis and therapeutic intervention.

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          Most cited references18

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          Risk of lymphoproliferative disorders after bone marrow transplantation: a multi-institutional study.

          G. Socié, G Sale, Jeffrey Curtis … (1999)
          We evaluated 18,014 patients who underwent allogeneic bone marrow transplantation (BMT) at 235 centers worldwide to examine the incidence of and risk factors for posttransplant lymphoproliferative disorders (PTLD). PTLD developed in 78 recipients, with 64 cases occurring less than 1 year after transplantation. The cumulative incidence of PTLD was 1.0% +/- 0.3% at 10 years. Incidence was highest 1 to 5 months posttransplant (120 cases/10,000 patients/yr) followed by a steep decline to less than 5/10,000/yr among >/=1-year survivors. In multivariate analyses, risk of early-onset PTLD ( /=3 major risk factors were 8.0% +/- 2.9% and 22% +/- 17.9%, respectively. We conclude that factors associated with altered immunity and T-cell regulatory mechanisms are predictors of both early- and late-onset PTLD.
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            Treatment of PTLD with rituximab or chemotherapy.

            D. P. Tsai, Rebecca Elstrom, Iona S. Schuster … (2006)
            Information regarding treatment of post-transplant lymphoproliferative disease (PTLD) beyond reduction in immunosuppression (RI) is limited. We retrospectively evaluated patients receiving rituximab and/or chemotherapy for PTLD for response, time to treatment failure (TTF) and overall survival (OS). Thirty-five patients met inclusion criteria. Twenty-two underwent rituximab treatment, with overall response rate (ORR) 68%. Median TTF was not reached at 19 months and estimated OS was 31 months. In univariable analysis, Epstein-Barr virus (EBV) positivity predicted response and TTF. LDH elevation predicted shorter OS. No patient died of rituximab toxicity and all patients who progressed underwent further treatment with chemotherapy. Twenty-three patients received chemotherapy. ORR was 74%, median TTF was 10.5 months and estimated OS was 42 months. Prognostic factors for response included stage, LDH and allograft involvement by tumor. These factors and lack of complete response (CR) predicted poor survival. Twenty-six percent of the patients receiving chemotherapy died of toxicity. Rituximab and chemotherapy are effective in patients with PTLD who fail or do not tolerate RI. While rituximab is well tolerated, toxicity of chemotherapy is marked. PTLD patients requiring therapy beyond RI should be considered for rituximab, especially with EBV-positive disease. Chemotherapy should be reserved for patients who fail rituximab, have EBV-negative tumors or need a rapid response.
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              Epstein-Barr virus-induced posttransplant lymphoproliferative disorders. ASTS/ASTP EBV-PTLD Task Force and The Mayo Clinic Organized International Consensus Development Meeting.

              C V Paya, J Fung, M Nalesnik … (1999)
              Epstein-Barr virus-induced posttransplant lymphoproliferative disease (EBV-PTLD) continues to be a major complication after solid organ transplantation in high-risk patients. Despite the identification of risk factors that predispose patients to develop EBV-PTLD, limitations in our knowledge of its pathogenesis, variable criteria for establishing the diagnosis, and lack of randomized studies addressing the prevention and treatment of EBV-PTLD hamper the optimal management of this transplant complication. This review summarizes the current knowledge of EBV-PTLD and, as a result of two separate international meetings on this topic, and provides recommendations for future areas of study.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Clin Med Case Rep
                Journal ID (iso-abbrev): Clin Med Case Rep
                Title: Clinical Medicine. Case Reports
                Publisher: Libertas Academica
                ISSN (Electronic): 1178-6450
                Publication date Collection: 2009
                Publication date (Electronic): 03 February 2009
                Volume: 2
                Pages: 11-15
                Affiliations
                [1 ]Université Denis Diderot—Paris 7, Assistance Publique-Hôpitaux de Paris, Service de Pneumologie, Hôpital Saint-Louis, Paris, France.
                [2 ]Université Denis Diderot—Paris 7, Assistance Publique-Hôpitaux de Paris, Service de Pathologie, Hôpital Saint-Louis, Paris, France.
                [3 ]Université Denis Diderot—Paris 7, Assistance Publique-Hôpitaux de Paris, Service d’Hématologie, Hôpital Saint-Louis, Paris, France.
                [4 ]Université Denis Diderot—Paris 7, Assistance Publique-Hôpitaux de Paris, Service de Greffe de moelle, Hôpital Saint-Louis, Paris, France.
                Author notes
                Correspondence: Anne Bergeron, Service de Pneumologie, Hôpital Saint-Louis, 1 Avenue Claude Vellefaux, 75010 Paris. Tel: (33)142494166; Fax: (33)142499395; Email: anne.bergeron-lafaurie@ 123456sls.aphp.fr
                Article
                Publisher ID: ccrep-2-2009-011
                PMC ID: 3785368
                PubMed ID: 24179366
                SO-VID: 36ee3017-fd14-4543-b1d4-f36fe5eba6fc
                Copyright © © 2009 by the authors
                License:

                This article is an open-access article distributed under the terms and conditions of the Creative Commons Attribution license ( http://creativecommons.org/licenses/by/3.0/).

                History
                Categories
                Subject: Case Report

                Keywords: bronchoscopy,endobronchial patterns,epstein-barr virus,hematopoietic stem cell transplantation,post transplant lymphoproliferative disorders
                Data availability:
                Keywords: bronchoscopy, endobronchial patterns, epstein-barr virus, hematopoietic stem cell transplantation, post transplant lymphoproliferative disorders

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