Disseminated BCG infection revealing a severe combined immunodeficiency: A case report

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Hajer Ben Belgacem
Jihene Bouguila
Amel Tej
Samia Tilouche
Raoudha Kebaili
Najoua Kahloul
Mohamed Ridha Barbouche
Nejla Soyah
Lamia Boughammoura

Abstract

Introduction: Bacillus Calmette Guerin (BCG) vaccine, which is administered to all newborns in Tunisia, can lead to serious complications
ranging from local disease to disseminated disease in a group of patients with primary immunodeficiency diseases.

Case report: A 3-month-old boy presented with persistent fever, hepato-splenomegaly and multiple osteolytic lesions. He was diagnosed
with severe combined immunodeficiency disease and disseminated BCG infection. Despite anti-tubercular therapy combined with intravenous
immunoglobulin, the evolution was fatal.

Conclusion: The case highlights the possible risk of such rare yet lethal complication of BCG vaccine. In suspected cases of primary immunodeficiency disease, inoculation of BCG should be postponed until appropriate screening tests exclude such diagnosis to prevent serious
complications.

Keywords:

Primary immunodeficiency, Mycobacterium Bovis, BCG vaccine

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References

  1. Sellami K, Amouri M, Kmiha S, Bahloul E, Aloulou H, Sfaihi L, et al. Adverse Reactions Due to the Bacillus CalmetteGuerin Vaccine: Twenty Tunisian Cases. Indian Journal of Dermatology. 2018; 63 (1):62 5
  2. Alfawaz TS, Alshehri M, Alshahrani D. BCG related complications: A single center, prospective observational study. International Journal of Pediatrics and Adolescent Medecine. 2015; 2(2):75 8
  3. Rezai MS, Khotaei G, Mamishi S, Kheirkhah M, Parvaneh N. Disseminated Bacillus Calmette-Guerin infection after BCG vaccination. Journal of Tropical Pediatrics. 2008; 54 (6):413 6
  4. Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D. The CARE Guidelines: Consensus-based Clinical Case Reporting Guideline Development. Global Advances Health Medecine. 2013;2 (5):38 43
  5. Sadeghi-Shabestari M, Rezaei N. Disseminated bacilleCalmetteGuérin in Iranian children with severe combined immunodeficiency. International Journal of Infectious Disease. 2009;13 (6): e420-423
  6. Barkai G, Somech R, Stauber T, Barziali A, Greenberger S. BacilleCalmette-Guerin (BCG) complications in children with severe combined immunodeficiency (SCID). Infectious Disease. 2019; 51(8):585 92
  7. Norouzi S, Aghamohammadi A, Mamishi S, Rosenzweig SD, Rezaei N. Bacillus Calmette-Guérin (BCG) complications associated with primary immunodeficiency diseases. Journal of Infection.2012; 64 (6):543 54.
  8. Sharma S, Tanigasalam V, Parameswaran N, Chandrasekaran V, Gulati R, Joseph NM, et al. Fulminant BCG Disease in a 7 Month Old Healthy Male Infant. Journal of Tropical Pediatrics.2015;61(6):474 7.
  9. Hassanzad M, Valinejadi A, Darougar S, Hashemitari SK, Velayati AA. Disseminated BacilleCalmette-Guérin infection at a glance: a mini review of the literature. Advances in respiratory medicine. 2019; 87 (4):239 42.
  10. Al-Mousa H. An infant with disseminated bacillus CalmetteGuerin infection (BCGitis) International Journal of Pediatrics and Adolescent Medicine.2014; 1(2): 89 92.
  11. Bernatowska E, Wolska-Kuœnierz B, Pac G, KurenkoDeptuch M, Pietrucha B, Zwolska Z, et al. Risk of BCG infection in primary immunodeficiency children. Proposal of diagnostic, prophylactic and therapeutic guidelines for disseminated BCG based on experience in the Department of Immunology, Children’s Memorial Health Institute in Warsaw between 1980 - 2006. Central European Journal of Immunology. 2007; 32 (4): 221-225.
  12. Huang LH, Shyur SD, Weng JD, Shin-Chi null, Tzen CY, Huang FY. Disseminated BacilleCalmette-Guérin disease as the initial presentation of X-linked severe combined immunodeficiency--a case report. Asian Pacific journal of allergy and immunology. 2005;23 (4):221 6.
  13. Culic S, Kuzmic I, Culic V, Martinic R, Kuljis D, Pranic-Kragic A, et al. Disseminated BCG infection resembling langerhans cell histiocytosis in an infant with severe combined immunodeficiency: a case report. Pediatric hematology and oncology.2004;21(6):563 72.
  14. Ben HadjHassine A, Marzouk M, Bargui H, Tfifha M, Dhaou M, Boukadida J. Disseminated BacilleCalmette-Guérin Infection in Immunodeficient Infants: Report of Two Cases. Archives of Pediatric Infectious Diseases.2016;5 (2).
  15. Aelami MH, Alborzi A, Pouladfar G, Geramizadeh B, Pourabbas B, Mardaneh J. Post-Vaccination Disseminated Bacillus Calmette Guerin Infection Among Children in Southern Iran. Jundishapur journal of microbiology.2015;8 (11):e25663.
  16. Lee PP. Disseminated Bacillus Calmette-Guérin and Susceptibility to Mycobacterial Infections-Implications on Bacillus Calmette-Guérin Vaccinations. Annals of the Academy of Medicine, Singapore.2015;44 (8):297 301.
  17. Shirvani F, Karimi A, Rajabnejad M. BCG Vaccination as a Prevention Strategy, Threats and Benefits. Archives of Pediatric Infectious Diseases. 2016;4 (2).
  18. Reljic R. IFN-gamma therapy of tuberculosis and related infections. Journal of interferon & cytokine research. 2007; 27(5):353-64
  19. Madkaikar M, Aluri J, Gupta S. Guidelines for Screening, Early Diagnosis and Management of Severe Combined Immunodeficiency (SCID) in India. Indian Journal Pediatrics. 2016;83(5):455 62.
  20. Marciano BE, Huang CY, Joshi G, Rezaei N, Carvalho BC, Allwood Z, et al. BCG vaccination in patients with severe combined immunodeficiency: complications, risks, and vaccination policies. The Journal of allergy and clinical immunology. 2014;133(4):1134 41.
  21. Al-Hammadi S, Alsuwaidi AR, Alshamsi ET, Ghatasheh GA, Souid AK. Disseminated Bacillus Calmette-Guérin (BCG) infections in infants with immunodeficiency. BMC research notes. 2017;10: 177.
  22. Bernatowska E, Skomska-Pawliszak M, Wolska-Kuśnierz B, Pac M, Heropolitanska-Pliszka E, Pietrucha B, et al. BCG Moreau Vaccine Safety Profile and NK CellsDouble Protection Against Disseminated BCG Infection in Retrospective Study of BCG Vaccination in 52 Polish Children with Severe Combined Immunodeficiency. Journal of clinical immunology. 2020;40(1):138 46.