Thoracic actinomycosis simulating neoplastic disease in Pediatrics

Abstracts

OBJETIVO: Descrever um caso de actinomicose pulmonar em paciente pediátrico. DESCRIÇÃO DO CASO: Menina de dez anos, saudável, com história de dor torácica há três meses, evoluindo há dois meses com abaulamento em parede torácica anterior esquerda. Ao exame físico, apresentava massa de 8,0cm de diâmetro em região inframamilar esquerda, sem sinais flogísticos. A tomografia do tórax mostrou processo expansivo em parede torácica anterior esquerda. Realizou-se biópsia e a cultura do material identificou Actinomyces israelli. Foi tratada com penicilina cristalina e amoxicilina, com desaparecimento da massa torácica. COMENTÁRIOS: A actinomicose é uma doença bacteriana rara, indolente, invasiva e, frequentemente, não considerada em crianças como causa provável de massas torácicas, sendo incomum o diagnóstico precoce.

actinomicose; tomografia; criança

CASE REPORT

Ricardo Mendes Pereira^I; Fabio Bucaretchi^I; Antonia Teresinha Tresoldi^II

Instituição: Departamento de Pediatria da Faculdade de Ciências Médicas da Universidade Estadual de Campinas (Unicamp), Campinas, SP, Brasil

^IDoutor em Saúde da Criança e do Adolescente pela Unicamp; Professor Doutor da Faculdade de Ciências Médicas da Unicamp, Campinas, SP, Brasil

^IIDoutora em Ciências Médicas pela Unicamp; Professora-Associada da Faculdade de Ciências Médicas da Unicamp, Campinas, SP, Brasil

Correspondence

ABSTRACT

OBJECTIVE: To describe a pulmonary actinomycosis case in a pediatric patient.

CASE DESCRIPTION: A ten-year-old healthy girl was admitted with a history of chest pain for the past three months. During the last two months, she evolved with bulging of the left anterior chest wall. Physical examination showed a mass with 8.0cm diameter in the left inframamilar region, without signs of inflammation. Chest tomography showed an expansion process in the left anterior chest wall. Biopsy and culture of the material identified Actinomyces israelli. She was treated with crystalline penicillin and amoxicillin, leading to the disappearance of the thoracic mass.

COMMENTS: Actinomycosis is a rare, indolent and invasive bacterial infection, and is often not considered in children as a cause of chest masses. Early diagnosis is uncommon.

Key-words: actinomycosis; actinomyces; tomography; child.

RESUMEN

OBJETIVO: Describir un caso de actinomicosis pulmonar en paciente pediátrico.

DESCRIPCIÓN DEL CASO: Muchacha de diez años, saludable, con historia de dolor torácico hace tres meses, evolucionando, hace dos meses, con abombamiento en pared torácica anterior izquierda. Al examen físico, presentaba masa de 8,0 cm de diámetro en región inframamilar izquierda, sin señales flogísticas. La tomografía del tórax mostró proceso expansivo en pared torácica anterior izquierda. Se realizó biopsia y la cultura del material identificó Actinomyces israelli. Se trató con penicilina cristalina y amoxicilina, con desaparición de la masa torácica.

COMENTARIOS: La actinomicosis es una enfermedad bacteriana rara, indolente, invasiva y, frecuentemente, no considerada en niños como causa probable de masas torácicas, siendo poco común el diagnóstico temprano.

Palabras clave: actinomicosis; actinomyces; tomografía; niño.

Introduction

Chest wall lesions in childhood include a wide range of pathologies; among the benign lesions, lipoma, neurofibroma, hemangioma, lynphangioma, and mesenchymal hamartoma stand out. Malignant lesions can be related to rhabdomyosarcoma and neuroblastoma. Manifestations of systemic diseases, such as leukemia, lymphoma, Langerhans cell histocytosis, and infections like tuberculosis and actinomycosis, may also cause chest wall lesions⁽¹⁾.

Thoracic actinomycosis is much more common in adults than in children and in male patients. Risk factors in children include dental caries, trauma, severe malnutrition, and uncontrolled diabetes mellitus^(2,3). We present the case of a ten-year old with pulmonary actinomycosis initially simulating malignant disease.

Case report

Ten-year-old female patient, previously healthy, was admitted with a history of continuous chest pain during the past three months, with a mass in the left anterior chest wall. In the last two months, she sought several health services, with no diagnosis, and was initially referred to a Pediatric Oncology Center with the hypothesis of malignant disease.

Physical examination showed a mass of nearly 8.0cm of diameter in the left inframammary region, hardened and with no signs of infection, as well as absence of caries or lymph node megaly and weight and height appropriate for age and sex. Laboratory tests showed: hemoglobin of 10.1g/dL, leukocyte count of 12,350/mm³ (66% of which were polymorphonuclear, 30% lymphocytes, and 4% monocytes), platelet count of 345,000/mm³, and high C-reactive protein (10.5mg/dL). Assessment of liver and kidney functions was normal. Tuberculin test was negative, and chest radiograph revealed a homogenous opacity in the upper left hemithorax, with increase in adjacent soft tissues (Figure 1). The contrast computed tomography scan showed a soft tissue mass in the left pulmonary lobe expanding to the left anterior mediastinum and the left anterior chest wall (Figure 2).

The anatomohistologic study revealed an inflammatory process with no evidence of malignancy. Hematoxylin-eosin (HE) staining revealed sulfur granules, suggesting Actinomyces. Investigation for alcohol-acid resistant bacilli was negative. Actinomyces israelii was identified in the culture of biopsy fragments (5% sheep's blood agar for anaerobic culture, Center for Disease Control).

The patient received crystalline penicillin (200,000units/kg/day) during three weeks, followed by six months of amoxicillin (40mg/kg/day). During the first week of treatment, she showed clinical and radiograph improvement. She was followed up every two weeks until completing oral antibiotic therapy and remained asymptomatic two years after the end of the treatment.

Discussion

Actinomycosis is an indolent, uncommon and invasive infection caused by several Actinomyces species, which are anaerobic facultative bacteria constituting the normal flora of oropharynx, intestinal tract, and female genital tract⁽²⁾. Nearly 20 Actinomyces species are known; however, A. israelii is the most frequently reported in human infections^(2,3). The most common sites involved are cervicofacial (60 to 40%), abdominopelvic (28 to 18%) and thoracic (34 to 14%) regions⁽²⁾.

Thoracic actinomycosis is a rare disease in childhood^(3-6). Bartlett et al⁽²⁾, in a literature review of studies conducted after computed tomography became available in 1975, identified 55 cases of thoracic actinomycosis in children and adolescents under 18 years old, mainly in boys (64%).

The most common findings in patients with thoracic actinomycosis are: chest wall mass (49%); pain (36%), generally located at the back of the elbow or at the axillary region; weight loss (35%); fever (35%); fistulas (15%); and hemoptysis (9%)^(2,3). Although some risk factors for infection have been described, such as the presence of dental caries, local trauma, foreign bodies, and uncontrolled diabetes^(2,4), their absence does not rule out the diagnosis of actinomycosis, as can be seen in the case described. Thoracic actinomycosis may be explained as a result of aspiration of Actinomyces and is also associated with tooth extractions.

The most frequent clinical presentation is chest wall mass, either mediastinal or pulmonary, as described in this case report. Pulmonary actinomycosis rarely presents as pneumonia, empyema or endobronchial mass^(2-8). Rib involvement is frequent, although sternum or vertebra invasion is rare^(2,4). Lower pulmonary lobes are the most affected⁽⁹⁾.

The diagnosis of actinomycosis is frequently delayed because it is not considered in the differential diagnosis for chest wall mass^(2,4); moreover, it is based on histology (sulfur granules). In the tissues, the bacteria develop into dense granules or microcolonies, called 'sulfur granules' due to its yellowish color, and may be observed on gross examination. In the series reported by Bartlett et al⁽²⁾, the diagnosis of actinomycosis was confirmed on histopathology in only 38% of patients.

Microorganisms of the Actinomyces genus are uniformly sensitive to penicillin, and the treatment is generally a combination of prolonged antibiotic therapy with surgical drainage. Despite the absence of available data to justify the optimal duration of treatment, most authors recommend that antibiotic treatment should last between six and 12 months, being administered intravenously at the beginning and then orally^(2,3,8-11). The evolution is satisfactory, rarely leading to death in children^(2,12).

The hypothesis of actinomycosis should always be considered in the differential diagnosis of chest masses, even in the absence of risk factors, which is important to guide microbiological and histological studies.

References

• 1. Watt AJ. Chest wall lesions. Paediatr Respir Rev 2002;3:328-38.
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• 3. Whitworth S, Jacobs RF. Actinomycosis. In: Feigin RD, Cherry J, Demmler-Harrison GJ, Kaplan SL, editors. Textbook of pediatric infectious diseases. 6^th ed. Philadelphia: Sanders; 2009. p. 1880-5.
• 4. Alborzi A, Pasyar N, Nasiri J. Actinomycosis as a neglected diagnosis of mediastinal mass. Jpn J Infect Dis 2006;59:52-3.
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• 9. Ganesan K, Khan R, Eisenhut M. Pulmonary actinomycosis masquerading as a malignant lung tumor in a 9-year-old boy. Clin Pediatr (Phila) 2005;44:181-3.
• 10. Teixeira AN, Alves CL, Martins Filho EP, Mendes EM, Nunes JT. Pulmonary actinomycosis simulating Tobias-Pancoast tumor. Rev Soc Bras Med Trop 2011;44:260-2.
• 11. Fatureto MC, Oliveira PF, Almeida CO, Fernandes LH. Lung actinomycosis with chest wall involvement. Rev Soc Bras Med Trop 2007;40:82-5.
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Thoracic actinomycosis simulating neoplastic disease in Pediatrics

Actinomicosis torácica simulando enfermedad neoplásica en Pediatría

Publication Dates

• Publication in this collection
  11 Oct 2012
• Date of issue
  Sept 2012

History

• Received
  03 Sept 2011
• Accepted
  12 Dec 2011