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Journal of Microbiology, Immunology,... Aug 2023
Review
Topics: Humans; Granulomatous Disease, Chronic; Hematopoietic Stem Cell Transplantation; Tuberculosis
PubMed: 36702645
DOI: 10.1016/j.jmii.2023.01.002 -
Asian Pacific Journal of Allergy and... Dec 2023Mendelian susceptibility to mycobacterial disease (MSMD) is a rare primary immunodeficiency, caused by non-tuberculous mycobacteria or Bacillus Calmette-Guerin (BCG)... (Review)
Review
BACKGROUND
Mendelian susceptibility to mycobacterial disease (MSMD) is a rare primary immunodeficiency, caused by non-tuberculous mycobacteria or Bacillus Calmette-Guerin (BCG) vaccine and characterized by severe diseases in childhood.
OBJECTIVE
In this study, we examined eight years followed-up 12 Turkish children with genetically proven MSMD and we tried to evaluate the survival rate with succesfull disease management, rate of consanguinity, molecular, cellular and clinical features of patients. In addition, we wanted to emphasize the importance of early diagnosis before administration of BCG vaccine in countries where this vaccine is routinely used.
METHODS
Twelve patients diagnosed with molecular studies [IFNγR1 complete (n = 1), IFNγR2 partial (n = 3), IL12Rβ1 (n = 6), NEMO (n = 1), STAT1 mutation (n = 1)] were included.
RESULTS
Ten patients (83%) were born from consanguineous parents and frequency of family history for the primary immunodeficiency was 58% (n = 7). All the cases had been immunized with BCG vaccine (Mycobacterium bovis) due to lack of early diagnosis. Two patients had BCG-itis and four patients had "BCG-osis". Survival rate was 75% after successful disease management with antibiotics, anti-tuberculous agents and recombinant IFN-γ.
CONCLUSIONS
It was concluded that MSMD must be differentiated from different forms of primary immunodeficiencies, so clinicians should be aware of MSMD especially in patients with BCG vaccine complications and non-tuberculous mycobacterial infection.
Topics: Humans; Child; BCG Vaccine; Follow-Up Studies; Mycobacterium Infections; Mycobacterium bovis; Mutation; Genetic Predisposition to Disease
PubMed: 33638623
DOI: 10.12932/AP-271219-0726 -
Journal of Clinical Immunology Oct 2023
Topics: Humans; Child; BCG Vaccine; COVID-19; Primary Immunodeficiency Diseases; STAT1 Transcription Factor
PubMed: 37258985
DOI: 10.1007/s10875-023-01510-x -
Arthroplasty Today Jun 2024Periprosthetic joint infection (PJI) can present challenges in diagnosis and treatment, particularly in the setting of atypical causative organisms such as fungi and...
Periprosthetic joint infection (PJI) can present challenges in diagnosis and treatment, particularly in the setting of atypical causative organisms such as fungi and mycobacteria. Herein, we present a case and provide a review of the diagnosis and treatment of an unusual PJI caused by bacillus Calmette-Guérin, administered during the treatment of bladder cancer 3 years prior to total knee arthroplasty and subsequent PJI. Although the patient's history of bladder cancer was known, neither his Bacillus Calmette-Guérin treatment nor its potential for distant site spread that could lead to PJI were appreciated, leading to a prolonged diagnostic evaluation and treatment course.
PubMed: 38533423
DOI: 10.1016/j.artd.2024.101350 -
Frontiers in Cellular and Infection... 2024(BCG) is a live strain of m () for use as an attenuated vaccine to prevent (TB) infection, while it could also lead to an infection in immunodeficient patients.... (Review)
Review
(BCG) is a live strain of m () for use as an attenuated vaccine to prevent (TB) infection, while it could also lead to an infection in immunodeficient patients. could infect patients with immunodeficiency via BCG vaccination. Disseminated BCG disease (BCGosis) is extremely rare and has a high mortality rate. This article presents a case of a 3-month-old patient with disseminated BCG infection who was initially diagnosed with hemophagocytic syndrome (HPS) and eventually found to have X-linked severe combined immunodeficiency (X-SCID). and its drug resistance genes were identified by metagenomics next-generation sequencing (mNGS) combined with targeted next-generation sequencing (tNGS) in blood and cerebrospinal fluid. Whole exome sequencing (WES) revealed a pathogenic variant in the common γ-chain gene (), confirming X-SCID. Finally, antituberculosis therapy and umbilical cord blood transplantation were given to the patient. He was successfully cured of BCGosis, and his immune function was restored. The mNGS combined with the tNGS provided effective methods for diagnosing rare BCG infections in children. Their combined application significantly improved the sensitivity and specificity of the detection of .
Topics: Male; Infant; Child; Humans; Mycobacterium bovis; BCG Vaccine; X-Linked Combined Immunodeficiency Diseases; Tuberculosis; Immunologic Deficiency Syndromes; Latent Tuberculosis; High-Throughput Nucleotide Sequencing
PubMed: 38410723
DOI: 10.3389/fcimb.2024.1341236 -
Journal of Medical Case Reports Jan 2024Intravesical Bacillus Calmette-Guérin (BCG) is used as a standard adjuvant therapy for non-muscle invasive urothelial cancer. Most patients tolerate the treatment...
BACKGROUND
Intravesical Bacillus Calmette-Guérin (BCG) is used as a standard adjuvant therapy for non-muscle invasive urothelial cancer. Most patients tolerate the treatment well, with mild side effects. Systemic complications are extremely rare, occur due to BCG dissemination and are associated with immunocompromised state and urothelial breach.
CASE PRESENTATION
We present a case of a 78-year-old male, a former smoker, with history of non-muscle invasive urothelial carcinoma status post partial resection followed by intravesical BCG therapy. An autopsy was performed due to the sudden nature of his death. Autopsy showed multiple necrotizing granulomas in the brain, atrium, ventricles, lungs, kidneys, and urinary bladder. Stains for acid-fast bacilli and fungi were negative. In addition, bilateral lungs showed evidence of bronchopneumonia secondary to cytomegalovirus.
CONCLUSION
Granulomatous myocarditis arising from BCG therapy is extremely rare. Our patient with urothelial cancer treated with BCG developed multiorgan granulomas, most likely due to a hypersensitivity reaction to intravesical BCG. Arrhythmia induced by granulomatous myocarditis was the cause of his death. Although there have been few cases of systemic BCG-osis causing fatal sepsis leading to death, a cardiac cause of death is unique.
Topics: Aged; Humans; Male; Autopsy; BCG Vaccine; Carcinoma, Transitional Cell; Granuloma; Myocarditis; Urinary Bladder Neoplasms; Fatal Outcome
PubMed: 38195538
DOI: 10.1186/s13256-023-04310-4