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      Acquired Pure Red Cell Aplasia Associated with Chronic Myelomonocytic Leukemia: Too Many of One, Not Enough of the Other

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          Abstract

          There is a growing body of literature outlining the association between certain hematological malignancies, such as chronic myelomonocytic leukemia (CMML), and systemic autoimmune diseases. Diagnosis and management can be difficult, particularly when autoimmune phenomena overlap with features of the underlying illness. This is especially the case in patients who develop immune-mediated cytopenias in the context of underlying bone marrow disease. CMML associated with immune thrombocytopenia and hemolytic anemia has been reported a number of times in the literature; however, there are only scattered case reports describing CMML associated with acquired pure red cell aplasia. Here, we describe the diagnostic and management approach to a patient who developed both diseases.

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          Most cited references 17

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          Systemic inflammatory and autoimmune manifestations associated with myelodysplastic syndromes and chronic myelomonocytic leukaemia: a French multicentre retrospective study.

          We describe myelodysplastic syndrome (MDS)-associated systemic inflammatory and autoimmune diseases (SIADs), their treatments and outcomes and the impact of SIADs on overall survival in a French multicentre retrospective study.
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            How I treat chronic myelomonocytic leukemia

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              Risk of myeloid neoplasms after solid organ transplantation

              Solid organ transplant recipients have elevated cancer risks, due in part to pharmacologic immunosuppression. However, little is known about risks for hematologic malignancies of myeloid origin. We linked the US Scientific Registry of Transplant Recipients with 15 population-based cancer registries to ascertain cancer occurrence among 207,859 solid organ transplants (1987–2009). Solid organ transplant recipients had significantly elevated risk for myeloid neoplasms, with standardized incidence ratios (SIRs) of 4.6 (95% confidence interval 3.8–5.6; N=101) for myelodysplastic syndromes (MDS), 2.7 (2.2–3.2; N=125) for acute myeloid leukemia (AML), 2.3 (1.6–3.2; N=36) for chronic myeloid leukemia, and 7.2 (5.4–9.3; N=57) for polycythemia vera. SIRs were highest among younger individuals and varied by time since transplantation and organ type (Poisson regression P<0.05 for all comparisons). Azathioprine for initial maintenance immunosuppression increased risk for MDS (P=0.0002) and AML (2–5 years after transplantation, P=0.0163). Overall survival following AML/MDS among transplant recipients was inferior to that of similar patients reported to US cancer registries (log-rank P<0.0001). Our novel finding of increased risks for specific myeloid neoplasms after solid organ transplantation supports a role for immune dysfunction in myeloid neoplasm etiology. The increased risks and inferior survival should heighten clinician awareness of myeloid neoplasms during follow-up of transplant recipients.
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                Author and article information

                Journal
                CRO
                CRO
                10.1159/issn.1662-6575
                Case Reports in Ophthalmology
                S. Karger AG
                1662-6575
                2020
                September - December 2020
                15 October 2020
                : 13
                : 3
                : 1270-1274
                Affiliations
                Department of Clinical Hematology, St Vincent’s Hospital, Melbourne, Victoria, Australia
                Author notes
                *Jose Filipe Gonsalves, Department of Clinical Hematology, St Vincent’s Hospital Melbourne, 41 Victoria Parade, Fitzroy, VIC (Australia), f.gonsalves@alfred.org.au
                Article
                508934 Case Rep Oncol 2020;13:1270–1274
                10.1159/000508934
                7670337
                © 2020 The Author(s). Published by S. Karger AG, Basel

                This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC). Usage and distribution for commercial purposes requires written permission. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                Page count
                Pages: 5
                Categories
                Case Report

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