614355
research-article2015
CPJXXX10.1177/0009922815614355Clinical PediatricsAcar et al
Brief Report
A Case of Empyema Necessitatis in a Child With Mycobacterium tuberculosis
Clinical Pediatrics 1–3 © The Author(s) 2015 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0009922815614355 cpj.sagepub.com
Manolya Acar, MD1, Murat Sutcu, MD1, Hacer Gundogdu Akturk, MD1, Ezgi Topyildiz, MD2, Ayper Somer, MD1, Oğuz Bulent Erol, MD3, Gonca Erkose Genc, MD4, and Nuran Salman, MD1 Introduction Empyema necessitatis (EN) is defined as extravasation of purulent material outside the pleural space involving the chest wall. The incidence of EN decreased significantly with appropriate antibiotic treatment, and it is rarely seen in the pediatric population.1,2 Mycobacterium tuberculosis is still the most common causative microorganism.3 Limited number of EN cases have also been reported due to infection with Actinomyces species, Streptococcus pneumonia, and Staphylococcus aureus.1,4 Herein we report a case of tuberculous EN in a child presenting as a chest wall mass.
Case Report An 11-year-old boy presented with a recent occurrence of swelling on his chest wall. That mass had appeared a week ago and he did not have any other complaints. His medical history was unremarkable until last year. He had been hospitalized with the complaint of cough, prolonged fever, and weight loss a year ago. His father had been diagnosed with cavitary tuberculosis at the same time. Chest X-ray showed massive plevral empyema on the left side. Tube thoracentesis had been performed and yielded exudative material. Acid-fast stain of the material was negative for M tuberculosis. Tuberculin skin test had been measured as 15 × 17 mm with positive BCG scar. Interferon-γ releasing test (IP-10) had been positive. Antituberculous therapy (isoniazide, rifampisin, pyrazinamide, ethambutol) was initiated and he was discharged with recovery. After that he did not come for regular follow-up. Retrospectively we found that he had refused to take his medication and plevral fluid culture had yielded M tuberculosis. On admission, inspection of the chest showed a nontender, fluctuant subcutaneous mass on the left side of anterior chest wall measuring 3 × 2 cm in diameter. Respiratory sounds were normal, and other system examinations were unremarkable. Initial laboratory evaluation including complete blood count, erythrocyte sedimentation rate, C-reactive protein, and routine blood chemistry were all within normal limits. Serum immunoglobulin
levels and lymphocyte subgroup analyses were normal. Anti-HIV antibody was also negative. Chest radiography showed an area of small infiltration in left lung parenchyma without plevral effusion (Figure 1). Superficial chest ultrasound revealed 36 × 24 mm of intense material collection on left anterior thorax wall that is passing through the wall and in communication with pleural cavity. Thorax computed tomography confirmed the extension of pleural material outside the thorax cavity. In addition, areas of plevral thickening and millimetric pulmonary nodules on both lungs were observed (Figure 2). Fine-needle aspiration of the chest wall abscess was performed. Acid-fast staining of the material revealed numerous acid-fast bacilli, and polymerase chain reaction for M tuberculosis revealed a positive result. The patient was restarted on antituberculosis therapy with isoniazid, rifampicin, pyrazinamide, and ethambutol since his previous plevral culture isolates were susceptible. The patient recovered well and was discharged from the hospital. Abscess disappeared with time. He is still being followed-up.
Discussion In the course of pulmonary tuberculosis, a small caseous focus in the lung parenchyma can rupture and leak into plevral cavity forming empyema. Without proper treatment, purulence in the plevral cavity may leak through surrounding soft tissue and extend outside. Since this is a chronic situation, slow growing, indolent microorganisms 1
Istanbul Medical Faculty, Pediatric Infectious Diseases, Istanbul University, Istanbul, Turkey 2 Istanbul Medical Faculty, Pediatrics, Istanbul University, Istanbul, Turkey 3 Istanbul Medical Faculty, Radiology, Istanbul University, Istanbul, Turkey 4 Istanbul Medical Faculty, Microbiology and Clinical Microbiology, Istanbul University, Istanbul, Turkey Corresponding Author: Manolya Acar, Department of Pediatric Infectious Diseases, Istanbul Medical Faculty, Istanbul University, Fatih/Capa, Istanbul 34093 Turkey. Email: [email protected]
Downloaded from cpj.sagepub.com at University of York on June 4, 2016
2
Clinical Pediatrics
Figure 1. Chest X ray with small infiltration on left side at the apical border.
Figure 2. Thorax computed tomography revealing extension of pleural material outside the thorax cavity.
like M tuberculosis are usually responsible. Our patient had been diagnosed as tuberculous empyema a year ago and given therapy but he refused to take his medication. One year later, he was presented with EN. Chest wall lesions in this situation may be defined as cold abscesses, since they are usually nonerythematous and nontender.3 Systemic signs of infections are usually absent. Our patient did not have any signs of systemic infection, and his initial laboratory workup including white blood cell count, erythrocyte sedimentation rate, and C-reactive protein were within normal limits. EN like other complications of empyema is more common in immunocompromised patients. But medical history and immunological evaluation of our patient revealed no abnormality. The incidence of EN has dropped with appropriate antibiotic treatment. Although Actinomycosis and Streptococcus species can be responsible, M tuberculosis still remains the most common causative microorganism.3
Freeman et al4 also identified Staphylococcus aureus, Fusobacterium nucleatum, and Mycobacterium avium intracellulare in their study. In occasional cases chest X-ray may not reveal any sign of EN. In our case, radiography of the chest showed only a small area of infiltration without plevral effusion. Diagnosis could be made with chest ultrasonography and computed tomography. In order to diagnose such cases, high levels of suspicion and further investigations may be necessary. Noninfectious etiologies should also be considered in differential diagnosis. Bronchogenic carcinoma, mesothelioma, and sarcomas are the most commonly observed primary pulmonary neoplasms, especially in the adult population. Although they are rare, Wegener’s granulomatosis, sarcoidosis, and infective endocarditis must be kept in mind.5 Empyema necessitatis can be very destructive. It may lead to erosion of bones and surrounding soft tissues. This may be asymptomatic at the beginning and progress in an indolent manner. Tuberculous EN treatment can be achieved with both surgical and medical interventions. Antituberculous therapy is initiated according to drug susceptibility. Recent data support the duration of antituberculous therapy between 6 months to a year.6 In our case, fortunately, the chest wall lesion was small and there was no destruction of bones. Because of this, surgical resection was not performed, and the lesion healed with antituberculosis treatment alone. In conclusion, pediatric EN is very uncommon. This may lead to underdiagnosis of pediatric cases in early stages. Morbidity and mortality can be high unless diagnosed early and appropriate treatment is given. Author Contributions MA, MS and ET were directly involved in the diagnosis and management of the case and were also responsible for the collection of clinical information and drafting of the manuscript. HGA was responsible for rigorous revision of the draft manuscript and collection of relevant references. OBE interpreted the radiological examination. GEG was responsible for microbiological analysis. AS and NS made critical revision of the final manuscript as submitted. Both authors read and approved the final manuscript.
Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
Downloaded from cpj.sagepub.com at University of York on June 4, 2016
3
Acar et al References 1. Tonna I, Conlon CP, Davies RJO. A case of empyema necessitatis. Eur J Intern Med. 2007;18:441-442. 2. Moore FO, Berne JD, McGovern TM, Ravishankar S, Slamon NB, Hertzog JH. Empyema necessitatis in an infant: a rare surgical disease. J Pediatr Surg. 2006;41:e.5-7. 3. Madeo J, Patel R, Gebre W, Ahmed S. Tuberculous empyema presenting as a persistent chest wall mass: case report. Germs. 2013;3:21-25.
4. Freeman AF, Ben-Ami T, Shulman ST. Streptococcus pneumonia empyema necessitatis. Pediatr Infect Dis J. 2004;23:177-178. 5. Kim Y, Lee SW, Choi HY, Im SA, Won T, Han WS. A case of pyothorax-associated lymphoma simulating empyema necessitatis. Clin Imaging. 2003;27:162-165. 6. Kim YT, Han KN, Kang CH, Sung SW, Kim JH. Complete resection is mandatory for tubercular cold abscess of the chest wall. Ann Thorac Surg. 2008;85:273-277.
Downloaded from cpj.sagepub.com at University of York on June 4, 2016
research-article2015
CPJXXX10.1177/0009922815614355Clinical PediatricsAcar et al
Brief Report
A Case of Empyema Necessitatis in a Child With Mycobacterium tuberculosis
Clinical Pediatrics 1–3 © The Author(s) 2015 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0009922815614355 cpj.sagepub.com
Manolya Acar, MD1, Murat Sutcu, MD1, Hacer Gundogdu Akturk, MD1, Ezgi Topyildiz, MD2, Ayper Somer, MD1, Oğuz Bulent Erol, MD3, Gonca Erkose Genc, MD4, and Nuran Salman, MD1 Introduction Empyema necessitatis (EN) is defined as extravasation of purulent material outside the pleural space involving the chest wall. The incidence of EN decreased significantly with appropriate antibiotic treatment, and it is rarely seen in the pediatric population.1,2 Mycobacterium tuberculosis is still the most common causative microorganism.3 Limited number of EN cases have also been reported due to infection with Actinomyces species, Streptococcus pneumonia, and Staphylococcus aureus.1,4 Herein we report a case of tuberculous EN in a child presenting as a chest wall mass.
Case Report An 11-year-old boy presented with a recent occurrence of swelling on his chest wall. That mass had appeared a week ago and he did not have any other complaints. His medical history was unremarkable until last year. He had been hospitalized with the complaint of cough, prolonged fever, and weight loss a year ago. His father had been diagnosed with cavitary tuberculosis at the same time. Chest X-ray showed massive plevral empyema on the left side. Tube thoracentesis had been performed and yielded exudative material. Acid-fast stain of the material was negative for M tuberculosis. Tuberculin skin test had been measured as 15 × 17 mm with positive BCG scar. Interferon-γ releasing test (IP-10) had been positive. Antituberculous therapy (isoniazide, rifampisin, pyrazinamide, ethambutol) was initiated and he was discharged with recovery. After that he did not come for regular follow-up. Retrospectively we found that he had refused to take his medication and plevral fluid culture had yielded M tuberculosis. On admission, inspection of the chest showed a nontender, fluctuant subcutaneous mass on the left side of anterior chest wall measuring 3 × 2 cm in diameter. Respiratory sounds were normal, and other system examinations were unremarkable. Initial laboratory evaluation including complete blood count, erythrocyte sedimentation rate, C-reactive protein, and routine blood chemistry were all within normal limits. Serum immunoglobulin
levels and lymphocyte subgroup analyses were normal. Anti-HIV antibody was also negative. Chest radiography showed an area of small infiltration in left lung parenchyma without plevral effusion (Figure 1). Superficial chest ultrasound revealed 36 × 24 mm of intense material collection on left anterior thorax wall that is passing through the wall and in communication with pleural cavity. Thorax computed tomography confirmed the extension of pleural material outside the thorax cavity. In addition, areas of plevral thickening and millimetric pulmonary nodules on both lungs were observed (Figure 2). Fine-needle aspiration of the chest wall abscess was performed. Acid-fast staining of the material revealed numerous acid-fast bacilli, and polymerase chain reaction for M tuberculosis revealed a positive result. The patient was restarted on antituberculosis therapy with isoniazid, rifampicin, pyrazinamide, and ethambutol since his previous plevral culture isolates were susceptible. The patient recovered well and was discharged from the hospital. Abscess disappeared with time. He is still being followed-up.
Discussion In the course of pulmonary tuberculosis, a small caseous focus in the lung parenchyma can rupture and leak into plevral cavity forming empyema. Without proper treatment, purulence in the plevral cavity may leak through surrounding soft tissue and extend outside. Since this is a chronic situation, slow growing, indolent microorganisms 1
Istanbul Medical Faculty, Pediatric Infectious Diseases, Istanbul University, Istanbul, Turkey 2 Istanbul Medical Faculty, Pediatrics, Istanbul University, Istanbul, Turkey 3 Istanbul Medical Faculty, Radiology, Istanbul University, Istanbul, Turkey 4 Istanbul Medical Faculty, Microbiology and Clinical Microbiology, Istanbul University, Istanbul, Turkey Corresponding Author: Manolya Acar, Department of Pediatric Infectious Diseases, Istanbul Medical Faculty, Istanbul University, Fatih/Capa, Istanbul 34093 Turkey. Email: [email protected]
Downloaded from cpj.sagepub.com at University of York on June 4, 2016
2
Clinical Pediatrics
Figure 1. Chest X ray with small infiltration on left side at the apical border.
Figure 2. Thorax computed tomography revealing extension of pleural material outside the thorax cavity.
like M tuberculosis are usually responsible. Our patient had been diagnosed as tuberculous empyema a year ago and given therapy but he refused to take his medication. One year later, he was presented with EN. Chest wall lesions in this situation may be defined as cold abscesses, since they are usually nonerythematous and nontender.3 Systemic signs of infections are usually absent. Our patient did not have any signs of systemic infection, and his initial laboratory workup including white blood cell count, erythrocyte sedimentation rate, and C-reactive protein were within normal limits. EN like other complications of empyema is more common in immunocompromised patients. But medical history and immunological evaluation of our patient revealed no abnormality. The incidence of EN has dropped with appropriate antibiotic treatment. Although Actinomycosis and Streptococcus species can be responsible, M tuberculosis still remains the most common causative microorganism.3
Freeman et al4 also identified Staphylococcus aureus, Fusobacterium nucleatum, and Mycobacterium avium intracellulare in their study. In occasional cases chest X-ray may not reveal any sign of EN. In our case, radiography of the chest showed only a small area of infiltration without plevral effusion. Diagnosis could be made with chest ultrasonography and computed tomography. In order to diagnose such cases, high levels of suspicion and further investigations may be necessary. Noninfectious etiologies should also be considered in differential diagnosis. Bronchogenic carcinoma, mesothelioma, and sarcomas are the most commonly observed primary pulmonary neoplasms, especially in the adult population. Although they are rare, Wegener’s granulomatosis, sarcoidosis, and infective endocarditis must be kept in mind.5 Empyema necessitatis can be very destructive. It may lead to erosion of bones and surrounding soft tissues. This may be asymptomatic at the beginning and progress in an indolent manner. Tuberculous EN treatment can be achieved with both surgical and medical interventions. Antituberculous therapy is initiated according to drug susceptibility. Recent data support the duration of antituberculous therapy between 6 months to a year.6 In our case, fortunately, the chest wall lesion was small and there was no destruction of bones. Because of this, surgical resection was not performed, and the lesion healed with antituberculosis treatment alone. In conclusion, pediatric EN is very uncommon. This may lead to underdiagnosis of pediatric cases in early stages. Morbidity and mortality can be high unless diagnosed early and appropriate treatment is given. Author Contributions MA, MS and ET were directly involved in the diagnosis and management of the case and were also responsible for the collection of clinical information and drafting of the manuscript. HGA was responsible for rigorous revision of the draft manuscript and collection of relevant references. OBE interpreted the radiological examination. GEG was responsible for microbiological analysis. AS and NS made critical revision of the final manuscript as submitted. Both authors read and approved the final manuscript.
Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
Downloaded from cpj.sagepub.com at University of York on June 4, 2016
3
Acar et al References 1. Tonna I, Conlon CP, Davies RJO. A case of empyema necessitatis. Eur J Intern Med. 2007;18:441-442. 2. Moore FO, Berne JD, McGovern TM, Ravishankar S, Slamon NB, Hertzog JH. Empyema necessitatis in an infant: a rare surgical disease. J Pediatr Surg. 2006;41:e.5-7. 3. Madeo J, Patel R, Gebre W, Ahmed S. Tuberculous empyema presenting as a persistent chest wall mass: case report. Germs. 2013;3:21-25.
4. Freeman AF, Ben-Ami T, Shulman ST. Streptococcus pneumonia empyema necessitatis. Pediatr Infect Dis J. 2004;23:177-178. 5. Kim Y, Lee SW, Choi HY, Im SA, Won T, Han WS. A case of pyothorax-associated lymphoma simulating empyema necessitatis. Clin Imaging. 2003;27:162-165. 6. Kim YT, Han KN, Kang CH, Sung SW, Kim JH. Complete resection is mandatory for tubercular cold abscess of the chest wall. Ann Thorac Surg. 2008;85:273-277.
Downloaded from cpj.sagepub.com at University of York on June 4, 2016
