A case of disseminated BCG infection in a daughter of Italian immigrants in Switzerland.

Bacillus Calmette-Guérin (BCG) is a vaccine against tuberculosis and contains a live, attenuated strain of Mycobacterium bovis as its essential constituent. Being a live, attenuated strain with potential pathogenicity, BCG can cause different complications, both near the inoculation site and through blood dissemination, especially in patients with immunodeficiency. IFN-γR1 deficiency is an autosomal recessively inherited immunodeficiency characterized by predisposition to infections with intracellular pathogens, in particular mycobacteria. We report a rare case of chronic osteomyelitis lasting 30 years due to BCG in a woman with IFN-γR1 deficiency who had previous clinical history of multi-organ BCGitis. Diagnosis of chronic osteomyelitis was confirmed by an 18-fluorine fluorodeoxyglucose positron emission tomography combined with CT scan (18F-FDG PET/CT). In children with a history of BCG vaccination and chronic unexplained infections, a clinical suspicion of BCG-related disease must arise, and a reason of immunodeficiency should be sought.

[1]  A. Cascio,et al.  Clinical use of BCG and its complications: a case series. , 2021, Le infezioni in medicina.

[2]  M. Netea,et al.  Licensed Bacille Calmette-Guérin (BCG) formulations differ markedly in bacterial viability, RNA content and innate immune activation , 2020, Vaccine.

[3]  Jian Lu,et al.  Clinical Spectrum of Complications Induced by Intravesical Immunotherapy of Bacillus Calmette-Guérin for Bladder Cancer , 2019, Journal of oncology.

[4]  M. Hassanzad,et al.  Disseminated Bacille Calmette-Guérin infection at a glance: a mini review of the literature. , 2019, Advances in respiratory medicine.

[5]  K. Alqurashi,et al.  Pediatric Disseminated Bacillus Calmette-Guerin Infection (BCGitis) : A Case Report , 2018 .

[6]  Ali Amanati,et al.  A 25-year surveillance of disseminated Bacillus Calmette–Guérin disease treatment in children in Southern Iran , 2017, Medicine.

[7]  P. Di Carlo,et al.  A Case of Epididymo-orchitis after intravesical bacille Calmette-Guérin therapy for superficial bladder carcinoma in a patient with latent tuberculosis infection , 2016, Infectious Agents and Cancer.

[8]  M. Breuning,et al.  Deletion of the entire interferon-γ receptor 1 gene causing complete deficiency in three related patients , 2016, Journal of Clinical Immunology.

[9]  L. Santos‐Argumedo,et al.  Innate defects of the IL-12/IFN-γ axis in susceptibility to infections by mycobacteria and salmonella. , 2014, Journal of interferon & cytokine research : the official journal of the International Society for Interferon and Cytokine Research.

[10]  Jason R Andrews,et al.  Timing of tuberculosis transmission and the impact of household contact tracing. An agent-based simulation model. , 2014, American journal of respiratory and critical care medicine.

[11]  G. Marks,et al.  Contact Tracing of Tuberculosis: A Systematic Review of Transmission Modelling Studies , 2013, PloS one.

[12]  C. Colomba,et al.  Disseminated tuberculosis in a patient treated with a JAK2 selective inhibitor: a case report , 2012, BMC Research Notes.

[13]  G. Marks,et al.  Contact investigation for tuberculosis: a systematic review and meta-analysis , 2012, European Respiratory Journal.

[14]  岡田 賢.  The novel IFNGR1 mutation 774del4 produces a truncated form of interferon-γ receptor 1 and has a dominant-negative effect on interferon-γ signal transduction , 2007 .

[15]  E. Bernatowska,et al.  Vaccination-related Mycobacterium bovis BCG Infection , 2006, Emerging infectious diseases.

[16]  M. Behr.  BCG--different strains, different vaccines? , 2002, The Lancet. Infectious diseases.

[17]  Capucine Picard,et al.  Impaired Interferon Gamma-Mediated Immunity and Susceptibility to Mycobacterial Infection in Childhood , 2001, Pediatric Research.

[18]  John Chan,et al.  Tuberculosis: Latency and Reactivation , 2001, Infection and Immunity.

[19]  M. Newport,et al.  A mutation in the interferon-gamma-receptor gene and susceptibility to mycobacterial infection. , 1996, The New England journal of medicine.

[20]  M. Newport,et al.  Interferon-gamma-receptor deficiency in an infant with fatal bacille Calmette-Guérin infection. , 1996, The New England journal of medicine.

[21]  C. Griscelli,et al.  Idiopathic disseminated bacillus Calmette-Guérin infection: a French national retrospective study. , 1996, Pediatrics.

[22]  A. Fischer,et al.  Immunological conditions of children with BCG disseminated infection , 1995, The Lancet.

[23]  J. Vilček,et al.  Mice That Lack the Interferon- 7 Receptor Have Profoundly Altered Responses to Infection with Bacillus Calmette-Gu6rin and Subsequent Ch-ilenge with Lipopolysaccharide By Ryutaro Kamijo,* Junming l.e,* Deborah Shapiro,* , 1993 .

[24]  O. Wasz‐Höckert,et al.  BCG complications. Estimates of the risks among vaccinated subjects and statistical analysis of their main characteristics. , 1984, Advances in tuberculosis research. Fortschritte der Tuberkuloseforschung. Progres de l'exploration de la tuberculose.