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Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological
Vincent Gies1,2,3, Yannick Dieudonné1,2, Florence Morel4,5
1Department 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.
Insights
Disseminated Bacillus Calmette-Guérin (BCG) infection is rare, typically seen in immunocompromised individuals. This case reveals a severe BCG infection in a patient with hematological malignancy, highlighting risks in secondary immunodeficiency.
Area of Science:
- Infectious Diseases
- Immunology
- Genetics
Background:
- Disseminated Mycobacterium bovis Bacillus Calmette-Guérin (BCG) infections are uncommon, primarily affecting individuals with inborn errors of immunity (IEI) or acquired immunodeficiency.
- While secondary BCG infections are documented following intravesical BCG instillation, this case presents a distinct scenario.
Observation:
- A 48-year-old male with a history of hematological malignancy developed severe disseminated BCG disease.
- The patient had no history of BCG instillation and no known IEI or defects in the IFNγ pathway.
- Whole genome sequencing identified the BCG strain as closely related to the M. bovis BCG Tice strain.
Findings:
- This case demonstrates a severe BCG infection in a patient with secondary immunodeficiency due to hematological malignancy.
- Genetic analysis of the BCG strain suggests a potential link between the patient's acquired immunodeficiency and the infection.
Implications:
- Physicians, microbiologists, and pharmacists must consider the risk of acquired disseminated BCG disease in immunocompromised patients.
- Awareness is crucial for patients undergoing BCG treatment for bladder cancer, especially those with secondary immunodeficiencies.
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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