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Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges
BACKGROUND: Lymphoproliferative disorder represents a heterogeneous clinicopathological spectrum characterized by uncontrolled proliferation of lymphocytes. Immunodeficiency is a major trigger of its development. While induction of immunodeficiency is a well-known adverse effect of temozolomide ther...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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BioMed Central
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10251676/ https://www.ncbi.nlm.nih.gov/pubmed/37296412 http://dx.doi.org/10.1186/s12883-023-03274-8 |
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author | Sato, Daisuke Takami, Hirokazu Takayanagi, Shunsaku Taoka, Kazuki Tanaka, Mariko Matsuura, Reiko Tanaka, Shota Saito, Nobuhito |
author_facet | Sato, Daisuke Takami, Hirokazu Takayanagi, Shunsaku Taoka, Kazuki Tanaka, Mariko Matsuura, Reiko Tanaka, Shota Saito, Nobuhito |
author_sort | Sato, Daisuke |
collection | PubMed |
description | BACKGROUND: Lymphoproliferative disorder represents a heterogeneous clinicopathological spectrum characterized by uncontrolled proliferation of lymphocytes. Immunodeficiency is a major trigger of its development. While induction of immunodeficiency is a well-known adverse effect of temozolomide therapy, development of lymphoproliferative disorder following temozolomide therapy has not previously been described. CASE PRESENTATION: A patient with brainstem glioma developed constitutional symptoms, pancytopenia, splenomegaly and generalized lymphadenopathy during the 2nd cycle of maintenance therapy following induction therapy with temozolomide. Epstein-Barr virus-infected lymphocytes were observed histopathologically and “other iatrogenic immunodeficiency-associated lymphoproliferative disorder” (OIIA-LPD) was diagnosed. Although discontinuation of temozolomide led to rapid remission, relapse was observed 4 months later. CHOP chemotherapy was induced, resulting in secondary remission. Vigilant follow-up for another 14 months showed radiologically stable brainstem glioma and no further recurrence of OIIA-LPD. CONCLUSIONS: This is the first report documenting OIIA-LPD during temozolomide administration. Timely diagnosis of the disease and discontinuation of the causative agent were considered to be the management of choice. Close monitoring for relapse should be continued. Finding a balance between glioma management and controlling the remission of OIIA-LPD remains to be clarified. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12883-023-03274-8. |
format | Online Article Text |
id | pubmed-10251676 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-102516762023-06-10 Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges Sato, Daisuke Takami, Hirokazu Takayanagi, Shunsaku Taoka, Kazuki Tanaka, Mariko Matsuura, Reiko Tanaka, Shota Saito, Nobuhito BMC Neurol Case Report BACKGROUND: Lymphoproliferative disorder represents a heterogeneous clinicopathological spectrum characterized by uncontrolled proliferation of lymphocytes. Immunodeficiency is a major trigger of its development. While induction of immunodeficiency is a well-known adverse effect of temozolomide therapy, development of lymphoproliferative disorder following temozolomide therapy has not previously been described. CASE PRESENTATION: A patient with brainstem glioma developed constitutional symptoms, pancytopenia, splenomegaly and generalized lymphadenopathy during the 2nd cycle of maintenance therapy following induction therapy with temozolomide. Epstein-Barr virus-infected lymphocytes were observed histopathologically and “other iatrogenic immunodeficiency-associated lymphoproliferative disorder” (OIIA-LPD) was diagnosed. Although discontinuation of temozolomide led to rapid remission, relapse was observed 4 months later. CHOP chemotherapy was induced, resulting in secondary remission. Vigilant follow-up for another 14 months showed radiologically stable brainstem glioma and no further recurrence of OIIA-LPD. CONCLUSIONS: This is the first report documenting OIIA-LPD during temozolomide administration. Timely diagnosis of the disease and discontinuation of the causative agent were considered to be the management of choice. Close monitoring for relapse should be continued. Finding a balance between glioma management and controlling the remission of OIIA-LPD remains to be clarified. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12883-023-03274-8. BioMed Central 2023-06-09 /pmc/articles/PMC10251676/ /pubmed/37296412 http://dx.doi.org/10.1186/s12883-023-03274-8 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data. |
spellingShingle | Case Report Sato, Daisuke Takami, Hirokazu Takayanagi, Shunsaku Taoka, Kazuki Tanaka, Mariko Matsuura, Reiko Tanaka, Shota Saito, Nobuhito Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
title | Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
title_full | Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
title_fullStr | Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
title_full_unstemmed | Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
title_short | Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
title_sort | lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10251676/ https://www.ncbi.nlm.nih.gov/pubmed/37296412 http://dx.doi.org/10.1186/s12883-023-03274-8 |
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