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High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review
BACKGROUND: Chimeric antigen receptor (CAR) T-cell therapy represents the most advanced immunotherapy against relapsed/refractory B cell malignancies. While cytokine release syndrome and immune effector cell-associated neurotoxicity syndrome are distinctive, known CAR T-cell acute adverse events, he...
Autores principales: | , , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2023
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9897055/ https://www.ncbi.nlm.nih.gov/pubmed/36741006 http://dx.doi.org/10.3389/fonc.2023.1036455 |
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author | Accorsi Buttini, Eugenia Farina, Mirko Lorenzi, Luisa Polverelli, Nicola Radici, Vera Morello, Enrico Colnaghi, Federica Almici, Camillo Ferrari, Emilio Bianchetti, Andrea Leoni, Alessandro Re, Federica Bosio, Katia Bernardi, Simona Malagola, Michele Re, Alessandro Russo, Domenico |
author_facet | Accorsi Buttini, Eugenia Farina, Mirko Lorenzi, Luisa Polverelli, Nicola Radici, Vera Morello, Enrico Colnaghi, Federica Almici, Camillo Ferrari, Emilio Bianchetti, Andrea Leoni, Alessandro Re, Federica Bosio, Katia Bernardi, Simona Malagola, Michele Re, Alessandro Russo, Domenico |
author_sort | Accorsi Buttini, Eugenia |
collection | PubMed |
description | BACKGROUND: Chimeric antigen receptor (CAR) T-cell therapy represents the most advanced immunotherapy against relapsed/refractory B cell malignancies. While cytokine release syndrome and immune effector cell-associated neurotoxicity syndrome are distinctive, known CAR T-cell acute adverse events, hematological toxicity has been increasingly reported. Cytopenia following CAR T-cell treatment is attributed in most cases to lymphodepletion regimens, bridging chemotherapy, or radiotherapy. However, when cytopenia becomes prolonged, the development of myelodysplastic syndrome (MDS) should be considered. CASE PRESENTATION: We report a case of high risk (HR)-MDS following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma. Eight months after CAR T-cell infusion, the blood count showed progressive, worsening cytopenia and the bone marrow biopsy revealed multilineage dysplasia without excess of blasts associated with chromosome 7 deletion and RUNX1 mutation. Next generation sequencing analysis, retrospectively performed on stored samples, showed a germ line CSF3R mutation, CEBPA clonal hematopoiesis, but no RUNX1 lesion. CONCLUSION: We describe a case of HR-MDS, with deletion of chromosome 7 and acquisition of RUNX1 mutation, developing after CAR T-cell therapy in a patient with clonal hematopoiesis (CH). Previous chemotherapy favored MDS onset; however, we could not exclude the fact that the impairment of immunosurveillance related to either lymphodepletion or CAR T-cell infusion may play a role in MDS development. Thus, we designed a multicenter prospective study (ClonHema-CAR-T-Study) to investigate if cytopenia after CAR T-cell treatment may be due to underling CH as well as the presence of secondary myeloid malignancies. |
format | Online Article Text |
id | pubmed-9897055 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-98970552023-02-04 High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review Accorsi Buttini, Eugenia Farina, Mirko Lorenzi, Luisa Polverelli, Nicola Radici, Vera Morello, Enrico Colnaghi, Federica Almici, Camillo Ferrari, Emilio Bianchetti, Andrea Leoni, Alessandro Re, Federica Bosio, Katia Bernardi, Simona Malagola, Michele Re, Alessandro Russo, Domenico Front Oncol Oncology BACKGROUND: Chimeric antigen receptor (CAR) T-cell therapy represents the most advanced immunotherapy against relapsed/refractory B cell malignancies. While cytokine release syndrome and immune effector cell-associated neurotoxicity syndrome are distinctive, known CAR T-cell acute adverse events, hematological toxicity has been increasingly reported. Cytopenia following CAR T-cell treatment is attributed in most cases to lymphodepletion regimens, bridging chemotherapy, or radiotherapy. However, when cytopenia becomes prolonged, the development of myelodysplastic syndrome (MDS) should be considered. CASE PRESENTATION: We report a case of high risk (HR)-MDS following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma. Eight months after CAR T-cell infusion, the blood count showed progressive, worsening cytopenia and the bone marrow biopsy revealed multilineage dysplasia without excess of blasts associated with chromosome 7 deletion and RUNX1 mutation. Next generation sequencing analysis, retrospectively performed on stored samples, showed a germ line CSF3R mutation, CEBPA clonal hematopoiesis, but no RUNX1 lesion. CONCLUSION: We describe a case of HR-MDS, with deletion of chromosome 7 and acquisition of RUNX1 mutation, developing after CAR T-cell therapy in a patient with clonal hematopoiesis (CH). Previous chemotherapy favored MDS onset; however, we could not exclude the fact that the impairment of immunosurveillance related to either lymphodepletion or CAR T-cell infusion may play a role in MDS development. Thus, we designed a multicenter prospective study (ClonHema-CAR-T-Study) to investigate if cytopenia after CAR T-cell treatment may be due to underling CH as well as the presence of secondary myeloid malignancies. Frontiers Media S.A. 2023-01-20 /pmc/articles/PMC9897055/ /pubmed/36741006 http://dx.doi.org/10.3389/fonc.2023.1036455 Text en Copyright © 2023 Accorsi Buttini, Farina, Lorenzi, Polverelli, Radici, Morello, Colnaghi, Almici, Ferrari, Bianchetti, Leoni, Re, Bosio, Bernardi, Malagola, Re and Russo https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Oncology Accorsi Buttini, Eugenia Farina, Mirko Lorenzi, Luisa Polverelli, Nicola Radici, Vera Morello, Enrico Colnaghi, Federica Almici, Camillo Ferrari, Emilio Bianchetti, Andrea Leoni, Alessandro Re, Federica Bosio, Katia Bernardi, Simona Malagola, Michele Re, Alessandro Russo, Domenico High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review |
title | High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review |
title_full | High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review |
title_fullStr | High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review |
title_full_unstemmed | High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review |
title_short | High risk-myelodysplastic syndrome following CAR T-cell therapy in a patient with relapsed diffuse large B cell lymphoma: A case report and literature review |
title_sort | high risk-myelodysplastic syndrome following car t-cell therapy in a patient with relapsed diffuse large b cell lymphoma: a case report and literature review |
topic | Oncology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9897055/ https://www.ncbi.nlm.nih.gov/pubmed/36741006 http://dx.doi.org/10.3389/fonc.2023.1036455 |
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