Grand Rounds

Is it BCGiosis or disseminated Tuberculosis?

1Seth G S Medical College, Mumbai, India, 2Pediatric Infectious Diseases, V Care Polyclinic, Mumbai, India

Address for Correspondence: Tsering Yangchen, Seth G S Medical College, Mumbai, India.

Keywords: Disseminated BCG, BCGiosis, immunocompetent

Clinical Problem :
An 11 months old girl presented with gradually increasing left axillary swelling for which she underwent an incision & drainage of the pus 1 week ago. Pus wall histology showed granulomatous inflammation suggestive of tuberculosis (TB). Smear did not show acid fast bacilli (AFB). She was started on anti TB treatment (ATT) consisting of Isoniazid (H), Rifampicin (R), Pyrazinamide (PZA), Ethambutol (E). She had received BCG at birth. Seven months ago, she was detected to have left frontal hemorrhage with subdural extension due to late hemorrhagic disease of the newborn. She underwent craniotomy and evacuation of the subdural hematoma. Subsequently 2 months after craniotomy, she was detected to have meningitis [cerebrospinal fluid proteins 144 gm%, 780 cells/cumm (80% polymorphs, 20% lymphocytes) and sugar 43 mg%] for which she was treated with intravenous (IV) antibiotics. On presentation to us, her weight was 6.7 kg, total length was 65 cm and she had a left axillary discharging sinus. There was a pea size swelling over extensor aspect of left wrist. X-ray of hand was normal and ultrasound abdomen was normal. After 1 month, the swelling over left wrist increased and there was a new swelling of left elbow. Pus was drained from left elbow that showed AFB on smear. She also developed hepatomegaly. Her ATT in form of HRZE was continued and additional drugs in form of amikacin, PAS, Ofloxacin, Clarithromycin and Ethionamide were added, pending culture and drug sensitivity report. Workup for immune deficiency in form of HIV, lymphocyte subset analysis (CD3, CD4, CD8, CD19 CD16/56) serum immunoglobulins were normal. TB culture at end of 6 weeks did not grow any organism. Six months after ATT, Amikacin was stopped. On completion of 1 year of ATT, her ATT was completely stopped. On her last follow up in April after 2 years of stopping ATT, she remained asymptomatic.

Is this BCGiosis or disseminated TB?

Discussion :
Bacillius Culmette Guerin (BCG) vaccine primarily used against TB is administered to all infants at high risk of TB immediately after birth. Its administration is often followed by mild side effects like redness, swelling and slight pain. Rarely, it may also be associated with serious complications, mainly in immunocompromised individuals which can be fatal.1,2,3,4 Certain cases have also been reported in individuals with competent immunity, which is even rarer.5,6 Complications may be benign, localized (BCGitis) or lethal disseminated (BCGiosis).7 It may probably be due to immature immune system of the infant which does not react effectively against the vaccine or may also be due to malnourishment which compromises immunity.5,6,8 Initially thought to be a disease of the infant, few cases have also been reported in older individuals, coinfected with HIV virus or in cases of revaccination of individuals who were did not react to the first vaccination.9 In a study conducted in Iran by Aelami et al, PCR has proven to be useful in the early and specific BCGiosis10 which is essential for initiating an anti-microbial therapy at the earliest. Mycobacterium bovis is inherently resistant to Pyrazinamide (PZA) and the use of HRE has found to be quite effective in its treatment.9 In our patient, we continued PZA as we could not confirm whether the infection was because of BCG or Mycobacterium tuberculosis (MTB) as we did not do a PCR test. However, with swelling starting in the left axilla in infancy and history of receiving BCG in the left deltoid pointed towards a BCGiosis. ATT combination of HRZ has been found to be effective in the presence of a competent immunity and its success rate in immunocompromised hosts is low. Similarly, our patient did not respond to HRZE initially and had dissemination of the disease. It was only after additional drugs were added that the infection resolved. We could not test for mendalian susceptibility for mycobacterial diseases (MSMD) due to non-affordability, however since the child had no further infections after stopping ATT, it seems to be unlikely to be MSMD.

References :
  1. Mohavali Z, Norouzi S, Mamishi S, Rezaei N. BCGiosis as a presenting feature of a child with chronic granulomatous disease. Braz J Infect Dis 2011;15:83-86.
  2. Norouzi S, Movahedi Z, Mamishi S, Monajemzadeh M, Razaei N. Disseminated BCG as a unique feature of an infant with severe combined immunodeficiency. Turk J Pediatr 2011;53:328-332.
  3. Hesseling AC, Marais BJ, Gie RP, Schaff HS, Fine PE, Godfrey -Fausseff P, et al. The risk of disseminated Bacille Calmette-Guerin [BCG] disease in HIV-infected children. Vaccine. 2007;25:14-18.
  4. Afshar Paiman S, Sidati A, Manushi S, Tabatabaie P, Khotaee G. Disseminated Mycobacterium bovis infection after BCG vaccination. Iran J Allergy Asthma Immunol. 2006;5:133-137
  5. Kido J, Mizukami T, Ohara O, Takada H, Yahai M. Idiopathic disseminated bacillus Calmette -Guerin infection in three infants. Pediatr Int. 2015;57:750-753.
  6. Casanova JL, Blanche S, Emile JF, Jouanguy E, Lamhamedis, Altare F, et al. Idiopathic Disseminated Bacillus Calmette-Guerin infection: A French National Retrospective Study. Pediatrics 1996;98:774-778.
  7. Kourime M, Akpalu EN, H Ouair, Jeddane L, Benhsaien I, Ailal F, et al. BCGitis/BCGiosis in children: Diagnosis, classification and treatment. Arch Pediatr. 2016;23:754-759.
  8. Trevenen CL, Pagtakhan RD. Disseminated tuberculoid lesion in infants following BCG vaccination. Can Med Assoc J. 1982;127:502-504.
  9. Talbot EA, Perkins MD, Silva SF, Frothingam R. Disseminated Bacillus Calmette-Guerin disease after vaccination: Case report and review. Clin Infec Dis. 1997;24:1139-1146.
  10. Aelami MH, Albourzi A, Pouladfar G, Geramizadeh B, Pourabbas B, Mardaneh J. Vaccinational Disseminated Bacillus Calmette Guerin Infection Among Children in Southern Iran. Jundishpur J Microbiol. 2015;8:e25663.

Correct Answers :  yes 100%
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