Sternal Osteomyelitis Associated with Bacillus Calmette–Guerin Moscow Strain
A
17-month-old immunocompetent boy presented with 2-month history of chest wall swelling. He had bacillus Calmette–Guerin (BCG) vaccination at 2 months of age and a doubtful tuberculosis contact history 5 months ago. He had a hard, fixed, 3 3 cm sternal swelling (Figure 1, A). Tuberculin skin test was negative. Chest radiograph did not reveal tuberculosis. Magnetic resonance imaging showed a destructive swollen mass on manubrium sterni with cystic necrotic areas (Figure 2; available at www. jpeds.com). Subsequently, the mass and destructive part of sternum were resected. Histopathologic examination revealed granulomatous inflammation, caseification, and acido-resistant bacilli. Tissue culture did not grow microorganism. Reverse transcription-polymerase chain reaction showed Mycobacterium tuberculosis complex. Because primary sternal tuberculosis osteomyelitis could not be excluded, antituberculosis chemotherapy (isoniazid, rifampicin, pyrazinamide, and ethambutol) was started. Subtype analysis (DNA sequencing and Hainlife Genotype MTBC kit; Nehren, Germany) identified descriptive bands for Mycobacterium bovis (1, 2, 3, 4, 7, 9, 10, and 13) Moscow strain, so pyrazinamide and ethambutol were discontinued. HIV serology and immunologic investigations were negative. At the end of 1-year treatment, complete recovery was achieved with no obvious complication (Figure 1, B). BCG is an old vaccine used in immunization programs in almost all countries. It is considered safe, and rarely leads to complications like osteomyelitis with an incidence between 0.39 and 369 per 1 million vaccination.1 As in this case, osteomyelitis generally occurs between 3 months to 5 years after vaccination.2 Moscow strain is a lesser known strain associated
with BCG complications. It is both challenging and important to discriminate between primary tuberculosis disease and BCG complication because of the differences in treatment. Whenever a BCG-related complication is suspected, subtype analysis is necessary for rapid diagnosis and appropriate therapy. n Soner Sertan Kara, MD Meltem Polat, MD Anil Tapisiz, MD Hasan Tezer, MD Pediatric Infectious Diseases Department
Meltem Yalinay Cirak, MD Microbiology Department
Cagri Damar, MD Radiology Department Gazi University Medical Faculty Ankara, Turkey
Cengiz Cavusoglu, MD Microbiology Department Ege University Medical Faculty Izmir, Turkey
References 1. Kim SH, Kim SY, Eun BW, Yoo WJ, Park KU, Choi EH, et al. BCG osteomyelitis caused by the BCG Tokyo strain and confirmed by molecular method. Vaccine 2008;26:4379-81. 2. Clavel G, Grados F, Lefauveau P, Fardellone P. Osteoarticular side effects of BCG therapy. Joint Bone Spine 2006;73:24-8.
Figure 1. A, The lesion at admission, and B, at the end of one year treatment.
J Pediatr 2015;-:---. 0022-3476/$ - see front matter. Copyright ª 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpeds.2015.02.040
1
THE JOURNAL OF PEDIATRICS
www.jpeds.com
Vol. -, No. -
Figure 2. A, Contrast enhanced T1-weighted fat suppressed axial, and B, contrast enhanced T1-weighted sagittal magnetic resonance images show a destructive swollen mass measuring 40 25 25 mm on the manubrium sterni and cartilage structures that is extending to the anterior mediastinum (arrows). It has centrally localized cystic necrotic areas and enhancement of surrounding soft tissues (A, arrow head).
1.e1
Kara et al
A
17-month-old immunocompetent boy presented with 2-month history of chest wall swelling. He had bacillus Calmette–Guerin (BCG) vaccination at 2 months of age and a doubtful tuberculosis contact history 5 months ago. He had a hard, fixed, 3 3 cm sternal swelling (Figure 1, A). Tuberculin skin test was negative. Chest radiograph did not reveal tuberculosis. Magnetic resonance imaging showed a destructive swollen mass on manubrium sterni with cystic necrotic areas (Figure 2; available at www. jpeds.com). Subsequently, the mass and destructive part of sternum were resected. Histopathologic examination revealed granulomatous inflammation, caseification, and acido-resistant bacilli. Tissue culture did not grow microorganism. Reverse transcription-polymerase chain reaction showed Mycobacterium tuberculosis complex. Because primary sternal tuberculosis osteomyelitis could not be excluded, antituberculosis chemotherapy (isoniazid, rifampicin, pyrazinamide, and ethambutol) was started. Subtype analysis (DNA sequencing and Hainlife Genotype MTBC kit; Nehren, Germany) identified descriptive bands for Mycobacterium bovis (1, 2, 3, 4, 7, 9, 10, and 13) Moscow strain, so pyrazinamide and ethambutol were discontinued. HIV serology and immunologic investigations were negative. At the end of 1-year treatment, complete recovery was achieved with no obvious complication (Figure 1, B). BCG is an old vaccine used in immunization programs in almost all countries. It is considered safe, and rarely leads to complications like osteomyelitis with an incidence between 0.39 and 369 per 1 million vaccination.1 As in this case, osteomyelitis generally occurs between 3 months to 5 years after vaccination.2 Moscow strain is a lesser known strain associated
with BCG complications. It is both challenging and important to discriminate between primary tuberculosis disease and BCG complication because of the differences in treatment. Whenever a BCG-related complication is suspected, subtype analysis is necessary for rapid diagnosis and appropriate therapy. n Soner Sertan Kara, MD Meltem Polat, MD Anil Tapisiz, MD Hasan Tezer, MD Pediatric Infectious Diseases Department
Meltem Yalinay Cirak, MD Microbiology Department
Cagri Damar, MD Radiology Department Gazi University Medical Faculty Ankara, Turkey
Cengiz Cavusoglu, MD Microbiology Department Ege University Medical Faculty Izmir, Turkey
References 1. Kim SH, Kim SY, Eun BW, Yoo WJ, Park KU, Choi EH, et al. BCG osteomyelitis caused by the BCG Tokyo strain and confirmed by molecular method. Vaccine 2008;26:4379-81. 2. Clavel G, Grados F, Lefauveau P, Fardellone P. Osteoarticular side effects of BCG therapy. Joint Bone Spine 2006;73:24-8.
Figure 1. A, The lesion at admission, and B, at the end of one year treatment.
J Pediatr 2015;-:---. 0022-3476/$ - see front matter. Copyright ª 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpeds.2015.02.040
1
THE JOURNAL OF PEDIATRICS
www.jpeds.com
Vol. -, No. -
Figure 2. A, Contrast enhanced T1-weighted fat suppressed axial, and B, contrast enhanced T1-weighted sagittal magnetic resonance images show a destructive swollen mass measuring 40 25 25 mm on the manubrium sterni and cartilage structures that is extending to the anterior mediastinum (arrows). It has centrally localized cystic necrotic areas and enhancement of surrounding soft tissues (A, arrow head).
1.e1
Kara et al
