TY - JOUR
T1 - Disseminated bacillus calmette-guérin lymphadenitis in a patient with gp91phox - Chronic granulomatous disease 25 years after vaccination
AU - Kusuhara, Koichi
AU - Ohga, Shouichi
AU - Hoshina, Takayuki
AU - Saito, Mitsumasa
AU - Sasaki, Yuka
AU - Ishimura, Masataka
AU - Takada, Hidetoshi
AU - Fujita, Masaki
AU - Hara, Toshiro
N1 - Funding Information:
Acknowledgments The authors thank Prof. Hiroyuki Nunoi (Department of Pediatrics, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan) for the genotyping of the patient with X-linked CGD and Dr. Masaaki Seki (Japan BCG Laboratory, Tokyo, Japan) for the identification of isolated mycobacterium. This work was supported in part by a Grant-in-Aid from the Ministry of Education, Culture, Sports, Science and Technology of Japan (no. 19591254) and Health and Labour Sciences Research Grants for Primary Immunodeficiency Diseases, for Tuberculosis, and for Vaccination from the Ministry of Health, Labour and Welfare of Japan.
PY - 2009/6
Y1 - 2009/6
N2 - A boy developed ipsilateral axillary lymphadenitis after Bacillus Calmette-Guérin (BCG) inoculation at the age of 5 months. Subsequently, he was diagnosed with X-linked chronic granulomatous disease (CGD) by the nitroblue tetrazolium assay when he was 4 years old. Body computerized tomography (CT) performed at the age of 25 years showed enlarged lymph nodes in the left periclavicular and axillary regions, and was confirmed by gallium scintigraphy. Mycobacterial culture, smear, and polymerase chain reaction (PCR) of the sputum and gastric fluid were negative. Whole-blood IFN-γ assay was negative as well. Mycobacterium bovis BCG was isolated from the lymph node biopsy by PCR amplification and culture. No mutation of the IFN-γ receptor 1 could be identified. In conclusion, CGD can be the underlying condition for BCG-itis; whole-blood IFN-γ assay might be useful in differentiating BCG infection and tuberculosis in CGD patients; BCG vaccination is contraindicated in X-linked CGD.
AB - A boy developed ipsilateral axillary lymphadenitis after Bacillus Calmette-Guérin (BCG) inoculation at the age of 5 months. Subsequently, he was diagnosed with X-linked chronic granulomatous disease (CGD) by the nitroblue tetrazolium assay when he was 4 years old. Body computerized tomography (CT) performed at the age of 25 years showed enlarged lymph nodes in the left periclavicular and axillary regions, and was confirmed by gallium scintigraphy. Mycobacterial culture, smear, and polymerase chain reaction (PCR) of the sputum and gastric fluid were negative. Whole-blood IFN-γ assay was negative as well. Mycobacterium bovis BCG was isolated from the lymph node biopsy by PCR amplification and culture. No mutation of the IFN-γ receptor 1 could be identified. In conclusion, CGD can be the underlying condition for BCG-itis; whole-blood IFN-γ assay might be useful in differentiating BCG infection and tuberculosis in CGD patients; BCG vaccination is contraindicated in X-linked CGD.
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U2 - 10.1007/s00431-008-0824-9
DO - 10.1007/s00431-008-0824-9
M3 - Article
C2 - 18762975
AN - SCOPUS:67349229164
SN - 0340-6199
VL - 168
SP - 745
EP - 747
JO - Acta Paediatrica Hungarica
JF - Acta Paediatrica Hungarica
IS - 6
ER -
