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Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection
Hydroa vacciniforme-like lymphoproliferative disorder (HV-LPD) is a cutaneous form of chronic active Epstein-Barrvirus (EBV) infection, which can develop into the extremely rare systemic lymphoma. Patients with Inborn errors of immunity (IEI), such as common variable immunodeficiency (CVID), are at...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9390486/ https://www.ncbi.nlm.nih.gov/pubmed/35990691 http://dx.doi.org/10.3389/fimmu.2022.915986 |
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author | Grześk, Elżbieta Kołtan, Sylwia Dąbrowska, Anna Urbańczyk, Anna Małdyk, Jadwiga Małkowski, Bogdan Bogiel, Tomasz Dębski, Robert Czyżewski, Krzysztof Wysocki, Mariusz Styczyński, Jan |
author_facet | Grześk, Elżbieta Kołtan, Sylwia Dąbrowska, Anna Urbańczyk, Anna Małdyk, Jadwiga Małkowski, Bogdan Bogiel, Tomasz Dębski, Robert Czyżewski, Krzysztof Wysocki, Mariusz Styczyński, Jan |
author_sort | Grześk, Elżbieta |
collection | PubMed |
description | Hydroa vacciniforme-like lymphoproliferative disorder (HV-LPD) is a cutaneous form of chronic active Epstein-Barrvirus (EBV) infection, which can develop into the extremely rare systemic lymphoma. Patients with Inborn errors of immunity (IEI), such as common variable immunodeficiency (CVID), are at higher risk of developing a severe course of infections especially viral and malignancies than the general population. The aim of the study was to present complex diagnostic and therapeutic management of HV-LPD. The clinical diagnosis was confirmed at the histological and molecular level with next generation sequencing. HV-LPD was diagnosed in a patient with CVID and chronic active Epstein–Barr virus (CAEBV) infection. The patient was refractory to CHOP chemotherapy and immunosuppressive treatment in combination with antiviral drugs (prednisone, bortezomib, gancyclovir). The third-party donor EBV-specific cytotoxic T cells (EBV-CTL, tabelecleucel) were used, which stabilised the disease course. Finally, matched unrelated donor hematopoietic cell transplantation (MUD-HCT) was performed followed by another cycle of EBV-CTL. |
format | Online Article Text |
id | pubmed-9390486 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-93904862022-08-20 Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection Grześk, Elżbieta Kołtan, Sylwia Dąbrowska, Anna Urbańczyk, Anna Małdyk, Jadwiga Małkowski, Bogdan Bogiel, Tomasz Dębski, Robert Czyżewski, Krzysztof Wysocki, Mariusz Styczyński, Jan Front Immunol Immunology Hydroa vacciniforme-like lymphoproliferative disorder (HV-LPD) is a cutaneous form of chronic active Epstein-Barrvirus (EBV) infection, which can develop into the extremely rare systemic lymphoma. Patients with Inborn errors of immunity (IEI), such as common variable immunodeficiency (CVID), are at higher risk of developing a severe course of infections especially viral and malignancies than the general population. The aim of the study was to present complex diagnostic and therapeutic management of HV-LPD. The clinical diagnosis was confirmed at the histological and molecular level with next generation sequencing. HV-LPD was diagnosed in a patient with CVID and chronic active Epstein–Barr virus (CAEBV) infection. The patient was refractory to CHOP chemotherapy and immunosuppressive treatment in combination with antiviral drugs (prednisone, bortezomib, gancyclovir). The third-party donor EBV-specific cytotoxic T cells (EBV-CTL, tabelecleucel) were used, which stabilised the disease course. Finally, matched unrelated donor hematopoietic cell transplantation (MUD-HCT) was performed followed by another cycle of EBV-CTL. Frontiers Media S.A. 2022-08-05 /pmc/articles/PMC9390486/ /pubmed/35990691 http://dx.doi.org/10.3389/fimmu.2022.915986 Text en Copyright © 2022 Grześk, Kołtan, Dąbrowska, Urbańczyk, Małdyk, Małkowski, Bogiel, Dębski, Czyżewski, Wysocki and Styczyński 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 | Immunology Grześk, Elżbieta Kołtan, Sylwia Dąbrowska, Anna Urbańczyk, Anna Małdyk, Jadwiga Małkowski, Bogdan Bogiel, Tomasz Dębski, Robert Czyżewski, Krzysztof Wysocki, Mariusz Styczyński, Jan Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection |
title | Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection |
title_full | Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection |
title_fullStr | Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection |
title_full_unstemmed | Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection |
title_short | Case report: Cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active Epstein-Barr virus infection |
title_sort | case report: cellular therapy for hydroa vacciniforme-like lymphoproliferative disorder in pediatric common variable immunodeficiency with chronic active epstein-barr virus infection |
topic | Immunology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9390486/ https://www.ncbi.nlm.nih.gov/pubmed/35990691 http://dx.doi.org/10.3389/fimmu.2022.915986 |
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