BCG-osis and Hematopoietic Cell Transplant for Primary Immunodeficiencies
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LETTER TO EDITOR
BCG-osis and Hematopoietic Cell Transplant for Primary Immunodeficiencies Anthony Sabulski 1,2
&
Stella M. Davies 1,2 & Grant Paulsen 1,3 & Rebecca Marsh 1,2 & Sharat Chandra 1,2
Received: 28 June 2020 / Accepted: 16 November 2020 # Springer Science+Business Media, LLC, part of Springer Nature 2020
To the Editor: Bacillus Calmette-Guérin (BCG) vaccination is an important global health measure in the prevention of Mycobacterium tuberculosis infection, but poses significant risks when administered to patients with primary immunodeficiencies (PIDs), in particular genetic defects associated with Mendelian Susceptibility to Mycobacterial Diseases (MSMD) or Severe Combined Immune Deficiency (SCID) [1]. BCG, a live vaccine, is contraindicated in patients with immune deficiency due to 400-fold and 33,000-fold increases in localized and disseminated BCG infections (BCG-osis) relative to the general population [1]. Many patients with SCID or MSMD require hematopoietic cell transplantation (HCT) for definitive therapy and limited data exist on the impact of BCG-osis and its associated therapies on HCT outcomes [2, 3]. The goals of our study were to investigate the effect of BCG-osis therapy on engraftment and determine the duration of BCG-osis therapy needed to prevent recurrence after HCT. We retrospectively reviewed records of six patients who underwent allogeneic HCT for PIDs complicated by BCGosis at Cincinnati Children’s Hospital Medical Center between 2013 and 2019. A total of 531 allogeneic HCTs were performed at our hospital during this time frame. As per criteria by Talbot et al. [4], disseminated BCG-osis was defined as a positive Mycobacterium bovis (BCG) culture and evidence of dissemination. Localized BCG-osis was defined as positive BCG culture at one anatomical site or evidence of
* Anthony Sabulski [email protected] 1
Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, USA
2
Cancer and Blood Diseases Institute, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Ave, Cincinnati, OH 45229, USA
3
Division of Infectious Diseases, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, USA
cellulitis, abscess, or localized lymphadenopathy at site of inoculation with or without positive BCG culture. Full donor chimerism was defined as whole blood donor chimerism > 90% and mixed chimerism defined as whole blood donor chimerism < 90%. Chimerism studies were done using either XY fluorescence in situ hybridization in the case of sex mismatched donor, or short-term tandem repeat analysis in the case of same sex donor. Acute and chronic GVHD were assessed and classified by standardized published criteria. All patients received standard supportive care and antimicrobial prophylaxis. Patient demographics and characteristics are shown in Table 1. Median age at HCT was 11.5 months (range 9– 23 months). Patients were born in the United Arab Emirates (n = 5) or Saudi Arabia (n = 1) and received the BCG vaccine at birth. Underlying diagnoses
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