Skip to main content

Advertisement

SpringerLink
Log in

Disseminated Bacillus Calmette-Guérin lymphadenitis in a patient with gp91phox − chronic granulomatous disease 25 years after vaccination

  • Short Report
  • Published:
European Journal of Pediatrics Aims and scope Submit manuscript

Abstract

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.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1

Abbreviations

CGD:

Chronic granulomatous disease

BCG:

Bacillus Calmette-Guérin

IFN:

Interferon

IL:

Interleukin

IFNGR1:

IFN-γ receptor 1

PID:

Primary immunodeficiency diseases

RD1:

Region of difference 1

ESAT-6:

Early secreted antigenic target 6-kDa protein

CFP-10:

10-kDa culture filtrate protein

References

  1. Bustamante J, Aksu G, Vogt G, de Beaucoudrey L, Genel F, Chapgier A, Filipe-Santos O, Feinberg J, Emile JF, Kutukculer N, Casanova JL (2007) BCG-osis and tuberculosis in a child with chronic granulomatous disease. J Allergy Clin Immunol 120:32–38

    Article  PubMed  Google Scholar 

  2. Filipe-Santos O, Bustamante J, Chapgier A, Vogt G, de Beaucoudrey L, Feinberg J, Jouanguy E, Boisson-Dupuis S, Fieschi C, Picard C, Casanova JL (2006) Inborn errors of IL-12/23- and IFN-gamma-mediated immunity: molecular, cellular, and clinical features. Semin Immunol 18:347–361

    Article  PubMed  CAS  Google Scholar 

  3. Lamhamedi-Cherradi S, de Chastellier C, Casanova JL (1999) Growth of Mycobacterium bovis, Bacille Calmette-Guérin, within human monocytes-macrophages cultured in serum-free medium. J Immunol Methods 225:75–86

    Article  PubMed  CAS  Google Scholar 

  4. Lee PP, Chan KW, Jiang L, Chen T, Li C, Lee TL, Mak PH, Fok SF, Yang X, Lau YL (2008) Susceptibility to mycobacterial infections in children with X-linked chronic granulomatous disease: a review of 17 patients living in a region endemic for tuberculosis. Pediatr Infect Dis J 27:224–230

    Article  PubMed  Google Scholar 

  5. Liese JG, Jendrossek V, Jansson A, Petropoulou T, Kloos S, Gahr M, Belohradsky BH (1996) Chronic granulomatous disease in adults. Lancet 347:220–223

    Article  PubMed  CAS  Google Scholar 

  6. Sasaki Y, Nomura A, Kusuhara K, Takada H, Ahmed S, Obinata K, Hamada K, Okimoto Y, Hara T (2002) Genetic basis of patients with bacille Calmette-Guérin osteomyelitis in Japan: identification of dominant partial interferon-gamma receptor 1 deficiency as a predominant type. J Infect Dis 185:706–709

    Article  PubMed  CAS  Google Scholar 

  7. Seki M, Sato A, Honda I, Yamazaki T, Yano I, Koyama A, Toida I (2005) Modified multiplex PCR for identification of Bacillus Calmette-Guérin substrain Tokyo among clinical isolates. Vaccine 23:3099–3102

    Article  PubMed  CAS  Google Scholar 

Download references

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.

Author information

Authors and Affiliations

  1. Department of Pediatrics, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka, 812-8582, Japan

    Koichi Kusuhara, Shouichi Ohga, Takayuki Hoshina, Mitsumasa Saito, Yuka Sasaki, Masataka Ishimura, Hidetoshi Takada & Toshiro Hara

  2. Department of Clinical Medicine, Research Institute for Diseases of the Chest, Kyushu University, Fukuoka, Japan

    Masaki Fujita

Authors
  1. Koichi Kusuhara
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Shouichi Ohga
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Takayuki Hoshina
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Mitsumasa Saito
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Yuka Sasaki
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Masataka Ishimura
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Hidetoshi Takada
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Masaki Fujita
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Toshiro Hara
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Shouichi Ohga.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Kusuhara, K., Ohga, S., Hoshina, T. et al. Disseminated Bacillus Calmette-Guérin lymphadenitis in a patient with gp91phox − chronic granulomatous disease 25 years after vaccination. Eur J Pediatr 168, 745–747 (2009). https://doi.org/10.1007/s00431-008-0824-9

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00431-008-0824-9

Keywords

Search

Navigation