Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy
Disseminated infections due to 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 B...
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| Published in | Frontiers in immunology Vol. 12; p. 696268 |
|---|---|
| Main Authors | , , , , , , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
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03.08.2021
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| Abstract | Disseminated infections due to
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.
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
BCG Tice strain, suggesting an unexpected relationship between the secondary immunodeficiency of the patient and the acquired BCG infection.
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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| AbstractList | 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.ContextDisseminated 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.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.Case DescriptionWe 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.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.ConclusionThis 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. Disseminated infections due to 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. 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 BCG Tice strain, suggesting an unexpected relationship between the secondary immunodeficiency of the patient and the acquired BCG infection. 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. ContextDisseminated 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 DescriptionWe 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.ConclusionThis 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. Context Disseminated infections due to Mycobacterium bovis Bacillus Calmette-Guerin (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. C ase 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 IFN gamma 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.& nbsp; 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. |
| Author | Korganow, Anne-Sophie Hubele, Fabrice Gies, Vincent Hansmann, Yves Danion, François Morel, Florence Carapito, Raphaël Jung, Sophie Kuhnert, Cornelia Dieudonné, Yannick Weingertner, Noëlle Schramm, Frederic Boyer, Pierre Guffroy, Aurélien Jacquel, Léa Sougakoff, Wladimir Martin, Aurélie |
| AuthorAffiliation | 7 Department of Infectiology, Strasbourg University Hospital , Strasbourg , France 6 Immunology Laboratory, Strasbourg University Hospital , Strasbourg , France 1 Department of Clinical Immunology and Internal Medicine, National Reference Center for Systemic Autoimmune Diseases (CNR RESO), Tertiary Center for Primary Immunodeficiency, Strasbourg University Hospital , Strasbourg , France 2 Université de Strasbourg, INSERM UMR-S1109, Institut thématique interdisciplinaire (ITI) de Médecine de Précision de Strasbourg, Transplantex NG, Faculté de médecine, Fédération Hospitalo-Universitaire OMICARE, Fédération de Médecine Translationnelle de Strasbourg (FMTS) , Strasbourg , France 4 APHP.Sorbonne Université, Hôpital Pitié-Salpêtrière, Laboratoire de Bactériologie-Hygiène, Centre National de Référence des Mycobactéries et de la Résistance des Mycobactéries aux Antituberculeux (CNR-MyRMA) , Paris , France 12 Laboratory of Bacteriology, Strasbourg University Hospital, Virulence bactérienne Précoce |
| AuthorAffiliation_xml | – name: 8 Departement of Pathology, Strasbourg University Hospital , Strasbourg , France – name: 2 Université de Strasbourg, INSERM UMR-S1109, Institut thématique interdisciplinaire (ITI) de Médecine de Précision de Strasbourg, Transplantex NG, Faculté de médecine, Fédération Hospitalo-Universitaire OMICARE, Fédération de Médecine Translationnelle de Strasbourg (FMTS) , Strasbourg , France – name: 11 Hôpitaux Universitaires de Strasbourg, Centre de Référence Maladies Rares Orales et Dentaires (O-Rares) , Pôle de Médecine et de Chirurgie Bucco-Dentaires, Strasbourg , France – name: 4 APHP.Sorbonne Université, Hôpital Pitié-Salpêtrière, Laboratoire de Bactériologie-Hygiène, Centre National de Référence des Mycobactéries et de la Résistance des Mycobactéries aux Antituberculeux (CNR-MyRMA) , Paris , France – name: 5 Sorbonne Universités, Inserm, Centre d'Immunologie et des Maladies Infectieuses (Cimi-Paris), UMR 1135 , Paris , France – name: 9 Departement of Nuclear Medicine and Molecular Imaging, ICANS, University Hospital of Strasbourg , Strasbourg , France – name: 10 Department of Internal Medicine, Strasbourg University Hospital , Strasbourg , France – name: 12 Laboratory of Bacteriology, Strasbourg University Hospital, Virulence bactérienne Précoce UR7290-Lyme Borreliosis Group, FMTS-CHRU Strasbourg, Institut de Bactériologie , Strasbourg , France – name: 3 Université de Strasbourg, Faculty of Pharmacy , Illkirch , France – name: 6 Immunology Laboratory, Strasbourg University Hospital , Strasbourg , France – name: 7 Department of Infectiology, Strasbourg University Hospital , Strasbourg , France – name: 1 Department of Clinical Immunology and Internal Medicine, National Reference Center for Systemic Autoimmune Diseases (CNR RESO), Tertiary Center for Primary Immunodeficiency, Strasbourg University Hospital , Strasbourg , France |
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| Cites_doi | 10.1155/2014/362845 10.1126/sciimmunol.aau8714 10.1056/NEJM199508313330905 10.1093/cid/cix496 10.1016/j.smim.2014.09.008 10.1093/clinids/14.3.662 10.1016/j.jaci.2008.10.037 10.1136/bcr-2016-215599 10.1007/s10875-019-00652-1 10.1016/j.cell.2013.09.045 10.1172/JCI19316 10.1016/j.medmal.2018.11.014 10.1086/498315 10.1007/s10875-020-00758-x 10.1016/j.jaip.2020.01.038 10.1080/20009666.2020.1742475 10.1016/S1473-3099(10)70196-1 10.1155/2013/821526 10.1086/313652 10.1056/NEJMoa062505 10.1097/MD.0000000000000119 10.1086/379034 |
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| Copyright | Copyright © 2021 Gies, Dieudonné, Morel, Sougakoff, Carapito, Martin, Weingertner, Jacquel, Hubele, Kuhnert, Jung, Schramm, Boyer, Hansmann, Danion, Korganow and Guffroy. Distributed under a Creative Commons Attribution 4.0 International License Copyright © 2021 Gies, Dieudonné, Morel, Sougakoff, Carapito, Martin, Weingertner, Jacquel, Hubele, Kuhnert, Jung, Schramm, Boyer, Hansmann, Danion, Korganow and Guffroy 2021 Gies, Dieudonné, Morel, Sougakoff, Carapito, Martin, Weingertner, Jacquel, Hubele, Kuhnert, Jung, Schramm, Boyer, Hansmann, Danion, Korganow and Guffroy |
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| Keywords | hematological malignancies case report BCGosis-susceptible PIDs BCG Immunodeficiency contamination |
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| License | Copyright © 2021 Gies, Dieudonné, Morel, Sougakoff, Carapito, Martin, Weingertner, Jacquel, Hubele, Kuhnert, Jung, Schramm, Boyer, Hansmann, Danion, Korganow and Guffroy. Distributed under a Creative Commons Attribution 4.0 International License: http://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. |
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| Notes | ObjectType-Case Study-2 SourceType-Scholarly Journals-1 ObjectType-Feature-4 content type line 23 ObjectType-Report-1 ObjectType-Article-3 Reviewed by: Saul Oswaldo Lugo Reyes, National Institute of Pediatrics, Mexico; Hassan Abolhassani, Karolinska University Hospital, Sweden Edited by: Sergio Rosenzweig, National Institutes of Health (NIH), United States This article was submitted to Primary Immunodeficiencies, a section of the journal Frontiers in Immunology |
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| Snippet | Disseminated infections due to
Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity (IEI) or acquired... Disseminated infections due to Mycobacterium bovis Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity (IEI)... Context Disseminated infections due to Mycobacterium bovis Bacillus Calmette-Guerin (BCG) are unusual and occur mostly in patients with inborn error of... ContextDisseminated infections due to Mycobacterium bovis Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity... |
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| SubjectTerms | Administration, Intravesical Antineoplastic Combined Chemotherapy Protocols - adverse effects Antineoplastic Combined Chemotherapy Protocols - therapeutic use Antitubercular Agents - therapeutic use BCG BCG Vaccine - administration & dosage BCG Vaccine - adverse effects BCGosis-susceptible PIDs case report contamination Hematologic Neoplasms - drug therapy hematological malignancies Host-Pathogen Interactions Human health and pathology Humans Immune Reconstitution Immunocompromised Host Immunodeficiency Immunology Life Sciences Male Middle Aged Mycobacterium bovis - drug effects Mycobacterium bovis - immunology Mycobacterium bovis - pathogenicity Opportunistic Infections - drug therapy Opportunistic Infections - immunology Opportunistic Infections - microbiology Risk Factors Treatment Outcome Tuberculosis, Pulmonary - diagnosis Tuberculosis, Pulmonary - drug therapy Tuberculosis, Pulmonary - immunology Tuberculosis, Pulmonary - microbiology Urinary Bladder Neoplasms - drug therapy Urinary Bladder Neoplasms - immunology |
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| Title | Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy |
| URI | https://www.ncbi.nlm.nih.gov/pubmed/34413849 https://www.proquest.com/docview/2563425995 https://hal.science/hal-03406526 https://pubmed.ncbi.nlm.nih.gov/PMC8369751 https://doaj.org/article/49b083e4653a42cb8a5e75877fc5d1cb |
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