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The Turkish Journal of Pediatrics 2016Pediatric sarcoidosis comprises a spectrum of childhood granulomatous inflammatory conditions. Pathological hallmark of the disease is granuloma formation that is seen...
Pediatric sarcoidosis comprises a spectrum of childhood granulomatous inflammatory conditions. Pathological hallmark of the disease is granuloma formation that is seen in the affected tissues and almost any organ or system can be involved. There are two forms of pediatric sarcoidosis. One is seen in older children and the clinical picture is very similar to that of adult sarcoidosis and the other one is seen in early childhood. Sarcoidosis in early childhood can be divided as Blau syndrome (familial form) and early onset sarcoidosis (sporadic form). In both of the diseases there is a defect in the NOD2/CARD15 gene. The typical triad of early onset sarcoidosis is polyarthritis, dermatitis and uveitis. Interferon-γ receptor 1 deficiency is caused by defects in the IFNγR1 gene and non-tuberculosis mycobacterial pathogens are the leading causes of infections that start in early childhood. Herein we report a patient who presented with the symptoms of early onset sarcoidosis and also had partial interferon-γ receptor 1 deficiency that presented with BCG-osis. In addition to anti-mycobacterial treatment, methotrexate and prednisolone were used in therapy.
Topics: Arthritis; BCG Vaccine; Humans; Infant; Interferon-gamma; Male; Receptors, Interferon; Sarcoidosis; Synovitis; Tomography, X-Ray Computed; Uveitis; Interferon gamma Receptor
PubMed: 28621099
DOI: 10.24953/turkjped.2016.05.015 -
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 -
The Journal of the Royal College of... Jun 2020Intra-vesical Bacillus Calmette-Guérin (BCG) immunotherapy is an effective treatment for high-risk bladder cancer. Less well known is that fewer than 1% of patients...
Intra-vesical Bacillus Calmette-Guérin (BCG) immunotherapy is an effective treatment for high-risk bladder cancer. Less well known is that fewer than 1% of patients receiving BCG treatment can develop disseminated BCG. The reaction can range from a mild flu-like illness to a systemic disorder with a fulminant course which in the most severe cases can lead to death. The diagnostic yield is low and diagnosis is often made after a comprehensive exclusion of more common causes of pyrexia of unknown origin. A high level of suspicion is therefore required in those who may be at risk. We report a case of disseminated BCG in an older patient for whom early involvement of his family was pertinent to determining the precipitant for delirium.
Topics: Aged; BCG Vaccine; Bacillus; Delirium; Humans; Treatment Outcome; Urinary Bladder Neoplasms
PubMed: 32568287
DOI: 10.4997/JRCPE.2020.215 -
Journal of Community Hospital Internal... May 2020Intravesical instillation of Bacillus-Calmette-Guerin (BCG), a live-attenuated-strain of , is an established treatment for superficial bladder carcinoma. Although...
Intravesical instillation of Bacillus-Calmette-Guerin (BCG), a live-attenuated-strain of , is an established treatment for superficial bladder carcinoma. Although generally well tolerated, 1/15,000 patients can develop life-threatening disseminated-BCG-infection typically soon after the procedure, a condition colloquially termed BCG-osis. Side-effects of intravesical BCG instillation including fever, chills, fatigue are common but BCG-osis is rare and severe, oftentimes requiring intensive care unit admission and triple anti-TB-therapy as in this case. It is therefore important for clinicians to recognize this possibility as the absence of specific signs and symptoms, coupled with the fastidious nature of the Mycobacteria, pose a diagnostic dilemma in the acute setting. Our case highlights this potential rare iatrogenic side effect of intravesical BCG treatment and the risk associated with non-treatment of BCG-osis.
PubMed: 32850058
DOI: 10.1080/20009666.2020.1742475 -
BMJ Case Reports Jul 2016A 69-year-old male patient who was treated with intravesical BCG for carcinoma in situ of the bladder, went on to develop systemic features of BCG-osis. This diagnosis...
A 69-year-old male patient who was treated with intravesical BCG for carcinoma in situ of the bladder, went on to develop systemic features of BCG-osis. This diagnosis was supported by significant radiological and clinical findings. These systemic features include pulmonary miliary lesions, a mycotic abdominal aortic aneurysm and penile lesions. Owing to a breakdown in the relationship between the patient and the National Health Service, the patient has declined BCG treatment. This case highlights the potential rare side effects of intravesical BCG treatment and the risk associated with non-treatment of BCG-osis.
Topics: Administration, Intravesical; Aged; Aneurysm, Infected; Animals; Aortic Aneurysm, Abdominal; BCG Vaccine; Carcinoma in Situ; Cattle; Granuloma; Humans; Male; Multiple Pulmonary Nodules; Penile Diseases; Tuberculosis, Bovine; Tuberculosis, Miliary; Urinary Bladder Neoplasms
PubMed: 27417990
DOI: 10.1136/bcr-2016-215635 -
BMJ Case Reports Dec 2019A 79-year-old man presented with an enlarging thoracic aneurysm on the background of superficial bladder cancer treated with intravesical bacillus Calmette-Guérin (BCG)...
A 79-year-old man presented with an enlarging thoracic aneurysm on the background of superficial bladder cancer treated with intravesical bacillus Calmette-Guérin (BCG) injections. Following the injections, he developed deranged liver function tests and hepatomegaly. Liver biopsy revealed granulomatous hepatitis compatible with disseminated mycobacterial infection (BCG-osis) and was treated with anti-tuberculosis agents for 12 months. A surveillance CT scan performed as a follow-up for his bladder cancer in 2018 revealed a saccular thoracic aneurysm at the ligamentum arteriosum, which was metabolically active on positron emission tomography (PET) scan. Given the timeframe from intravesical instillation of BCG and the metabolic activity on PET scan, the lesion was consistent with a mycotic aneurysm secondary to disseminated mycobacterial infection. Following multidisciplinary team discussion, a thoracic endovascular aneurysm repair was performed. The stent grafts were placed distal to the left subclavian artery with good angiographic results and no immediate postoperative complications. He was initiated on long-term antibiotics to cover potential bacterial pathogens including mycobacterium.
Topics: Adjuvants, Immunologic; Aged; Animals; Anti-Bacterial Agents; Antitubercular Agents; Aorta, Thoracic; Aortic Aneurysm, Abdominal; BCG Vaccine; Humans; Male; Mycobacterium Infections; Positron Emission Tomography Computed Tomography; Urinary Bladder Neoplasms
PubMed: 31843771
DOI: 10.1136/bcr-2019-231595 -
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 -
MMWR. Morbidity and Mortality Weekly... Aug 2014Poliovirus transmission has been eliminated in most of the world through the use of inactivated poliovirus vaccine (IPV) and live, attenuated oral poliovirus vaccine...
Poliovirus transmission has been eliminated in most of the world through the use of inactivated poliovirus vaccine (IPV) and live, attenuated oral poliovirus vaccine (OPV). In the United States, use of OPV was discontinued by the year 2000 because of the potential for vaccine-associated paralytic polio (VAPP); an average of eight cases were reported each year in the United States during 1980-2000. Polio eradication efforts in other parts of the world continue to rely on OPV to take advantage of transmission of poliovirus vaccine strains to unvaccinated persons in the population, lower cost, and ease of administration. In 2013, an infant aged 7 months who recently immigrated to the United States from India was referred to a hospital in San Antonio, Texas. The infant had fever, an enlarging skin lesion in the deltoid region with axillary lymphadenopathy, decreased activity, and inability to bear weight on the left leg, progressing to paralysis of the left leg over a 6-week period. Recognition of lymphopenia on complete blood count led to immune evaluation, which revealed the presence of severe combined immunodeficiency syndrome (SCIDS), an inherited disorder. A history of OPV and bacille Calmette-Guérin (BCG) vaccination in India led to the diagnoses of VAPP and BCG-osis, which were confirmed microbiologically. This report demonstrates the importance of obtaining a comprehensive clinical history in a child who has recently immigrated to the United States, with recognition that differing vaccine practices in other countries might require additional consideration of potential etiologies.
Topics: BCG Vaccine; Emigrants and Immigrants; Fatal Outcome; Humans; India; Infant; Male; Paralysis; Poliomyelitis; Poliovirus Vaccine, Oral; Severe Combined Immunodeficiency; Texas; Tuberculosis; Vaccines, Attenuated
PubMed: 25144542
DOI: No ID Found