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Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy

CONTEXT: Disseminated infections due to Mycobacterium bovis Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity (IEI) or acquired immunodeficiency. However, cases of secondary BCGosis due to intravesical BCG instillation have been described. Herein,...

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Autores principales: Gies, Vincent, Dieudonné, Yannick, Morel, Florence, Sougakoff, Wladimir, Carapito, Raphaël, Martin, Aurélie, Weingertner, Noëlle, Jacquel, Léa, Hubele, Fabrice, Kuhnert, Cornelia, Jung, Sophie, Schramm, Frederic, Boyer, Pierre, Hansmann, Yves, Danion, François, Korganow, Anne-Sophie, Guffroy, Aurélien
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8369751/
https://www.ncbi.nlm.nih.gov/pubmed/34413849
http://dx.doi.org/10.3389/fimmu.2021.696268
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author Gies, Vincent
Dieudonné, Yannick
Morel, Florence
Sougakoff, Wladimir
Carapito, Raphaël
Martin, Aurélie
Weingertner, Noëlle
Jacquel, Léa
Hubele, Fabrice
Kuhnert, Cornelia
Jung, Sophie
Schramm, Frederic
Boyer, Pierre
Hansmann, Yves
Danion, François
Korganow, Anne-Sophie
Guffroy, Aurélien
author_facet Gies, Vincent
Dieudonné, Yannick
Morel, Florence
Sougakoff, Wladimir
Carapito, Raphaël
Martin, Aurélie
Weingertner, Noëlle
Jacquel, Léa
Hubele, Fabrice
Kuhnert, Cornelia
Jung, Sophie
Schramm, Frederic
Boyer, Pierre
Hansmann, Yves
Danion, François
Korganow, Anne-Sophie
Guffroy, Aurélien
author_sort Gies, Vincent
collection PubMed
description CONTEXT: Disseminated infections due to Mycobacterium bovis Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity (IEI) or acquired immunodeficiency. However, cases of secondary BCGosis due to intravesical BCG instillation have been described. Herein, we present a case of severe BCGosis occurring in an unusual situation. CASE DESCRIPTION: We report one case of severe disseminated BCG disease occurring after hematological malignancy in a 48-year-old man without BCG instillation and previously vaccinated in infancy with no complication. Laboratory investigations demonstrated that he was not affected by any known or candidate gene of IEI or intrinsic cellular defect involving IFNγ pathway. Whole genome sequencing of the BCG strain showed that it was most closely related to the M. bovis BCG Tice strain, suggesting an unexpected relationship between the secondary immunodeficiency of the patient and the acquired BCG infection. CONCLUSION: This case highlights the fact that, in addition to the IEI, physicians, as well as microbiologists and pharmacists should be aware of possible acquired disseminated BCG disease in secondary immunocompromised patients treated in centers that administrate BCG for bladder cancers.
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spelling pubmed-83697512021-08-18 Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy Gies, Vincent Dieudonné, Yannick Morel, Florence Sougakoff, Wladimir Carapito, Raphaël Martin, Aurélie Weingertner, Noëlle Jacquel, Léa Hubele, Fabrice Kuhnert, Cornelia Jung, Sophie Schramm, Frederic Boyer, Pierre Hansmann, Yves Danion, François Korganow, Anne-Sophie Guffroy, Aurélien Front Immunol Immunology CONTEXT: Disseminated infections due to Mycobacterium bovis Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity (IEI) or acquired immunodeficiency. However, cases of secondary BCGosis due to intravesical BCG instillation have been described. Herein, we present a case of severe BCGosis occurring in an unusual situation. CASE DESCRIPTION: We report one case of severe disseminated BCG disease occurring after hematological malignancy in a 48-year-old man without BCG instillation and previously vaccinated in infancy with no complication. Laboratory investigations demonstrated that he was not affected by any known or candidate gene of IEI or intrinsic cellular defect involving IFNγ pathway. Whole genome sequencing of the BCG strain showed that it was most closely related to the M. bovis BCG Tice strain, suggesting an unexpected relationship between the secondary immunodeficiency of the patient and the acquired BCG infection. CONCLUSION: This case highlights the fact that, in addition to the IEI, physicians, as well as microbiologists and pharmacists should be aware of possible acquired disseminated BCG disease in secondary immunocompromised patients treated in centers that administrate BCG for bladder cancers. Frontiers Media S.A. 2021-08-03 /pmc/articles/PMC8369751/ /pubmed/34413849 http://dx.doi.org/10.3389/fimmu.2021.696268 Text en Copyright © 2021 Gies, Dieudonné, Morel, Sougakoff, Carapito, Martin, Weingertner, Jacquel, Hubele, Kuhnert, Jung, Schramm, Boyer, Hansmann, Danion, Korganow and Guffroy 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
Gies, Vincent
Dieudonné, Yannick
Morel, Florence
Sougakoff, Wladimir
Carapito, Raphaël
Martin, Aurélie
Weingertner, Noëlle
Jacquel, Léa
Hubele, Fabrice
Kuhnert, Cornelia
Jung, Sophie
Schramm, Frederic
Boyer, Pierre
Hansmann, Yves
Danion, François
Korganow, Anne-Sophie
Guffroy, Aurélien
Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
title Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
title_full Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
title_fullStr Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
title_full_unstemmed Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
title_short Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
title_sort case report: acquired disseminated bcg in the context of a delayed immune reconstitution after hematological malignancy
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8369751/
https://www.ncbi.nlm.nih.gov/pubmed/34413849
http://dx.doi.org/10.3389/fimmu.2021.696268
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