BCG vaccine
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Omenn’s syndrome with disseminated BCGosis in an infant: case report A male infant developed Omenn’s syndrome with disseminated BCGosis following immunisation with BCG vaccine [route and dosage not stated]. The boy presented at 8 months of age. His symptoms had started at 1 month of age, after BCG vaccination. They included fever, erythematous skin rash, scalp abscesses, lymphadenopathy, hepatosplenomegaly, cough and failure to thrive. He had a large non-healed ulcer at the vaccination site. Laboratory investigations included the following: platelet count 401 × 103/µL, WBC count 6.5 × 103/µL, lymphocyte count 2.2 × 103/µL, neutrophil count 2.3 × 103/µL, total bilirubin 5.8 mg/dL, direct bilirubin 3.6 mg/dL, GGT 147 U/L, IgG 71 mg/dL, IgM 13.4 mg/dL, CD19 0.14 and eosinophilia. Swab culture from the ulcer revealed the presence of Staphylococcus epidermidis, and a single blood culture showed pseudomonas infection. Diffuse bilateral infiltrates were seen on a chest x-ray. A diagnosis of Omenn’s syndrome (T+, B-, NK+) with disseminated BCGosis was made. The boy received immune globulin, dexamethasone, cotrimoxazole [sulfamethoxazole/trimethoprim], fluconazole, aciclovir, isoniazid, rifampicin, ethambutol, ciprofloxacin, amikacin and clarithromycin. His condition improved and, 4 weeks later, he underwent allogenic bone marrow transplantation (BMT). Eight days after the transplant, he developed a flare of local BCGosis with left axillary lymphadenitis, followed by an erythematous skin rash considered to be grade 1 skin graft-versus-host disease (GVHD). He received methylprednisolone, and was discharged on day +32 post-BMT. He was readmitted on day +49 with bone marrow GVHD and pancytopenia. He was retransplanted from the same donor on day +87. Forty days after the second transplant, he was admitted with grade 3 skin GVHD and hepatic involvement. On day +90, he developed a mild flare of local BCGosis which required adjustment of antituberculosis treatment and the addition of amikacin. At last follow-up, he was in a stable condition. Wahadneh A, et al. Successful second bone marrow transplantation in Omenn’s syndrome after bone marrow aplasia: A case report. Pediatric Transplantation 16: E43-E48, No. 2, Mar 2012. Available from: URL: http://dx.doi.org/10.1111/ 803070452 j.1399-3046.2010.01413.x - Jordan
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Reactions 19 May 2012 No. 1402
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