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Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia

Idiopathic acquired aplastic anemia (AA) is a rare lymphocyte-mediated bone marrow aplasia. No specific mechanisms or triggers of AA have been identified. We recently observed several patients who developed AA after Hodgkin lymphoma (HL). We hypothesized that the co-occurrence of HL and AA is not ra...

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Autores principales: Linaburg, Taylor, Davis, Adam R., Frey, Noelle V., Khawaja, Muhammad R., Landsburg, Daniel J., Schuster, Stephen J., Svoboda, Jakub, Li, Yimei, Borovskiy, Yuliya, Olson, Timothy S., Bagg, Adam, Hexner, Elizabeth O., Babushok, Daria V.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6450628/
https://www.ncbi.nlm.nih.gov/pubmed/30951562
http://dx.doi.org/10.1371/journal.pone.0215021
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author Linaburg, Taylor
Davis, Adam R.
Frey, Noelle V.
Khawaja, Muhammad R.
Landsburg, Daniel J.
Schuster, Stephen J.
Svoboda, Jakub
Li, Yimei
Borovskiy, Yuliya
Olson, Timothy S.
Bagg, Adam
Hexner, Elizabeth O.
Babushok, Daria V.
author_facet Linaburg, Taylor
Davis, Adam R.
Frey, Noelle V.
Khawaja, Muhammad R.
Landsburg, Daniel J.
Schuster, Stephen J.
Svoboda, Jakub
Li, Yimei
Borovskiy, Yuliya
Olson, Timothy S.
Bagg, Adam
Hexner, Elizabeth O.
Babushok, Daria V.
author_sort Linaburg, Taylor
collection PubMed
description Idiopathic acquired aplastic anemia (AA) is a rare lymphocyte-mediated bone marrow aplasia. No specific mechanisms or triggers of AA have been identified. We recently observed several patients who developed AA after Hodgkin lymphoma (HL). We hypothesized that the co-occurrence of HL and AA is not random and may be etiologically significant. To test this hypothesis, we determined the incidence of AA in HL patients at our institution. We identified four patients with co-occurring HL and AA, with the incidence of AA in HL patients >20-fold higher compared to the general population. We identified 12 additional patients with AA and HL through a systematic literature review. Of the 16 total patients,15 (93.8%) developed AA after or concurrent with a HL diagnosis. None of the patients had marrow involvement by HL. Five of 15 patients were in complete remission from HL at the time of AA diagnosis, and six had a concurrent presentation with no prior cytotoxic therapy, with diagnostic timeframe information unavailable for four patients. The median interval between HL diagnosis and AA onset was 16 months, ranging from concurrent to 14 years after a HL diagnosis. The median survival after AA diagnosis was 14 months (range: 1 month to 20 years). Our results show that patients with HL have a higher incidence of AA compared to the general population and suggest that HL-related immune dysregulation may be a risk factor for AA. Better recognition and management of AA in HL patients is needed to improve outcomes in this population.
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spelling pubmed-64506282019-04-19 Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia Linaburg, Taylor Davis, Adam R. Frey, Noelle V. Khawaja, Muhammad R. Landsburg, Daniel J. Schuster, Stephen J. Svoboda, Jakub Li, Yimei Borovskiy, Yuliya Olson, Timothy S. Bagg, Adam Hexner, Elizabeth O. Babushok, Daria V. PLoS One Research Article Idiopathic acquired aplastic anemia (AA) is a rare lymphocyte-mediated bone marrow aplasia. No specific mechanisms or triggers of AA have been identified. We recently observed several patients who developed AA after Hodgkin lymphoma (HL). We hypothesized that the co-occurrence of HL and AA is not random and may be etiologically significant. To test this hypothesis, we determined the incidence of AA in HL patients at our institution. We identified four patients with co-occurring HL and AA, with the incidence of AA in HL patients >20-fold higher compared to the general population. We identified 12 additional patients with AA and HL through a systematic literature review. Of the 16 total patients,15 (93.8%) developed AA after or concurrent with a HL diagnosis. None of the patients had marrow involvement by HL. Five of 15 patients were in complete remission from HL at the time of AA diagnosis, and six had a concurrent presentation with no prior cytotoxic therapy, with diagnostic timeframe information unavailable for four patients. The median interval between HL diagnosis and AA onset was 16 months, ranging from concurrent to 14 years after a HL diagnosis. The median survival after AA diagnosis was 14 months (range: 1 month to 20 years). Our results show that patients with HL have a higher incidence of AA compared to the general population and suggest that HL-related immune dysregulation may be a risk factor for AA. Better recognition and management of AA in HL patients is needed to improve outcomes in this population. Public Library of Science 2019-04-05 /pmc/articles/PMC6450628/ /pubmed/30951562 http://dx.doi.org/10.1371/journal.pone.0215021 Text en © 2019 Linaburg et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Article
Linaburg, Taylor
Davis, Adam R.
Frey, Noelle V.
Khawaja, Muhammad R.
Landsburg, Daniel J.
Schuster, Stephen J.
Svoboda, Jakub
Li, Yimei
Borovskiy, Yuliya
Olson, Timothy S.
Bagg, Adam
Hexner, Elizabeth O.
Babushok, Daria V.
Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
title Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
title_full Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
title_fullStr Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
title_full_unstemmed Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
title_short Hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
title_sort hodgkin lymphoma patients have an increased incidence of idiopathic acquired aplastic anemia
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6450628/
https://www.ncbi.nlm.nih.gov/pubmed/30951562
http://dx.doi.org/10.1371/journal.pone.0215021
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