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Journal of Indian Association of Pediatric Surgeons
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Table of Contents   
CASE REPORT
Year : 2023  |  Volume : 28  |  Issue : 6  |  Page : 526-528
 

Pulmonary actinomycosis in children: A prompt diagnosis can prevent surgery


Department of General Surgery, Division of Paediatric Surgery, King Hamad University Hospital, Busaiteen, Bahrain

Date of Submission21-Apr-2023
Date of Decision03-Jul-2023
Date of Acceptance03-Aug-2023
Date of Web Publication02-Nov-2023

Correspondence Address:
Prince Raj
King Hamad University Hospital, Building 2345, Road 2835, Block 228, P. O. Box 24343, Busaiteen
Bahrain
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.jiaps_86_23

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   Abstract 


Pulmonary actinomycosis is an extremely rare disease in children. The diagnosis is challenging as the clinical presentation and radiological investigations may be atypical. We report a case of a pulmonary lesion extending to the chest wall posing as a malignant lesion. Biopsy revealed colonies of Actinomyces. Antibiotic therapy resolved the mass. A rare diagnosis of pulmonary actinomycosis should be kept as a differential diagnosis of such a mass in children.


Keywords: Actinomycosis, infection, pulmonary, sarcoma


How to cite this article:
Raj P, Shankar R, Alawadi MA, Corbally M. Pulmonary actinomycosis in children: A prompt diagnosis can prevent surgery. J Indian Assoc Pediatr Surg 2023;28:526-8

How to cite this URL:
Raj P, Shankar R, Alawadi MA, Corbally M. Pulmonary actinomycosis in children: A prompt diagnosis can prevent surgery. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Nov 28];28:526-8. Available from: https://www.jiaps.com/text.asp?2023/28/6/526/389332





   Introduction Top


Pulmonary actinomycosis is a rare chronic infection caused by Actinomyces, a Gram-positive, microaerophilic bacterium. It is a commensal in the mouth and gastrointestinal and vaginal tracts. Pulmonary involvement is often misdiagnosed as pulmonary tuberculosis, lung abscess, or even lung tumor. Infection may cause cavities, nodules, consolidations, and effusion; hence, even with clinical suspicion, it can lead to a diagnostic dilemma. In particular, when soft tissue is involved, it can be confused with sarcoma. Early diagnosis is prudent as it will avoid unnecessary psychological and physical morbidities involving surgery, and it helps in starting early antibiotic therapy, which is curative.[1]


   Case Report Top


A 6-year-old girl presented with complaints of a painless swelling in the posterior aspect of her right chest for 1 week. She had a history of a fall 10 days before it. The swelling gradually increased in size over the next few days with an episode of fever. There was no history of cough or recent contact with coronavirus disease (COVID-19). The mother gave a history of weight loss and night sweats in recent months. General physical examination was unremarkable except for significant dental caries [Figure 1]. Local examination revealed a 5 cm × 4 cm nontender firm swelling, just inferior to the tip of the right scapula, diffuse margins, no increased temperature, and the skin overlying the swelling was unremarkable. In view of the above clinical findings, a chest wall tumor/organized hematoma/inflammatory lesion was kept as a differential diagnosis in that order.
Figure 1: Clinical picture showing dental caries

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The hematological investigation showed a hemoglobin of 12.3 g/dl, WBC of 10,000/μL, C-reactive protein of 10, and erythrocyte sedimentation rate of 120 mm/1st h. Chest X-ray and ultrasound showed an ill-defined opacity involving the upper and mid-zone of the right lung with significant soft-tissue involvement of the adjacent chest wall. A magnetic resonance imaging (MRI) of the thorax [Figure 2]a showed a subcutaneous soft-tissue lesion (15 mm × 43 mm × 55 mm) in the right posterolateral aspect of the thoracic wall extending between the rib and into the pleural space. There was no destruction or increase in the distance between the ribs and the right scapula. There was no intraforaminal extension or spinal involvement. Consolidation was noted in the right upper lobe posterior segment, the posterior part of the middle lobe, and the whole lower lobe. Bronchoscopy was not considered in this child as we thought of this lesion as a primarily extrapulmonary lesion involving the lung, and she did not have any cough or expectoration. In view of the MRI findings, and a possibility of a malignant mass lesion, a tru-cut biopsy was done and reported as granulation tissue with marked inflammatory infiltrate. No granuloma or malignancy was noted. Immunohistochemical studies reported smooth muscle actin positive for myofibroblasts. CD68 was positive for histiocytes. Pan-cytokeratin (AE1/AE3) and ALK stains were negative.
Figure 2: (a) Magnetic resonance imaging coronal views a subcutaneous soft-tissue lesion (15 mm × 43 mm × 55 mm) in the right posterolateral aspect of the thoracic wall extending between the rib and into the pleural space. Consolidation noted in the right upper lobe posterior segment, posterior part of the middle lobe, and whole lower lobe, (b) Colonies of Actinomyces and no evidence of malignancy

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Positron emission tomography (PET)–computed tomography (CT) and an open biopsy were done in view of inconclusive core biopsy and high suspicion of malignancy (peripheral neuroectodermal tumor, Ewing's sarcoma, and lymphoma). The PET-CT showed hypermetabolic right posterior hemithorax mass encasing the posterior parts of multiple ribs with the intra- and extrathoracic soft-tissue component, where the involved ribs showed lamellated periosteal reaction associated with hypermetabolic lytic changes further strengthening our suspicion of a malignant lesion.

However, an open biopsy of the extrathoracic soft tissue component relieved us, as it showed colonies of Actinomyces and no evidence of malignancy [Figure 2]b.

The child was started on intravenous (IV) penicillin G 2.5 Mu six hourly, and she responded well. IV antibiotics were continued for 3 weeks, and she was later switched to oral amoxicillin 500 mg thrice daily for the next 6 months. Chest X-ray done at the end of 10 days of IV therapy showed marked improvement with clinical resolution of the chest wall mass. Her dental treatment was with the removal of caries teeth.

She is completely asymptomatic and has returned to her normal daily routine at 1 year of follow-up. Chest X-ray at 1 year shows no abnormality.


   Discussion Top


Pulmonary actinomycosis is a rare type of infection in children, which is insidious in onset and progressive over time. Although it is often reported in adult populations with male predominance, there are a few reports of pediatric pulmonary actinomycosis. The infection is caused by anaerobic Gram-positive bacteria from Actinomyces species, initially misclassified in the 19th century as fungi.[1] Actinomyces israelii is the most common isolated pathogen, although Actinomyces meyeri is also frequently encountered in cases of pulmonary involvement. These organisms are normally found in the human oral cavity, and the disease usually develops as a result of aspiration of these pathogens from oropharyngeal or gastrointestinal secretions. The pathology of pulmonary actinomycosis probably stems when saliva laden with Actinomyces species is aspirated into a terminal bronchus, causing atelectasis and pneumonitis.[2] The initial acute inflammation is followed by a characteristic chronic, indolent phase that generates local necrosis and fibrosis and commonly cavitates. It progresses with little regard for anatomic boundaries eventually crossing the interlobar fissures. If unchecked, the infection invades the pleura, chest wall, soft tissues, and bony structures; sinus tracts may form, opening and closing spontaneously.

Children with dental caries, similar to our case, are particularly predisposed to develop these infections. The frequency of this disease is higher in those individuals with underlying immunodeficiency, malnutrition, and following radiotherapy. Due to its rarity, nonspecific clinical presentation, and imaging, pulmonary Actinomyces is often not entertained as a differential diagnosis. Hence, diagnosis may be difficult and significantly delayed. Symptomatology may include cough with purulent sputum (mostly in adults), weight loss, low-grade fever, weakness, and breathlessness, although none of these were present in our patient except for weight loss. Marked weight loss, malaise, and high fever may be a pointer of disseminated disease.[3] Occasionally, there may be a history of hemoptysis and chest pain. Although now, it is a rare infection with a very low mortality rate,[4] early accurate diagnosis will prevent the considerable morbidity, psychological and physical, associated with either delayed or missed diagnosis.

Misdiagnosis, particularly as a malignancy, is distressing for the patient who may end up with a lung resection for essentially a benign disease curable completely with antibiotics. In a case series, the diagnosis of pulmonary actinomycosis was suspected on admission in only 7% of patients who later proved to have the infection.[5] The average duration of illness before definitive diagnosis was 6 months, a consistent figure in most cases. Even when the clinical suspicion is high, microbiological confirmation can still be difficult. Communication between the surgeon and the microbiologist, collection techniques, and delivery of tissue specimens in anaerobic culture media are vital. The infection shares many clinical features with chronic lung infections, such as tuberculosis, fungal infections, and also lung malignancy. Up to a quarter of cases of thoracic actinomycosis are initially diagnosed as malignancy. Finding Actinomyces filaments in sputum alone, particularly without the characteristic sulfur granules, may therefore represent colonization of the devitalized tissue. Thus, short of exploratory thoracotomy and differentiation from lung carcinoma may sometimes be impossible. The diagnosis, therefore, requires a combination of several factors, including a positive culture and demonstration of sulfur granules in purulent matter from infected tissue, correlation with the clinical and radiological features, and the response to antibiotic treatment. In our case, the diagnosis was challenging as neither the history nor the clinical presentations gave a clue to the diagnosis and even the radiology, including MRI and PET-CT were more in favor of malignancy. The final diagnosis was achieved only on open biopsy. We would stress on adequate tissue biopsy for better histopathological and immunocytochemical analyses.

The prognosis of the pulmonary form of actinomycosis may be less favorable compared with the other more common forms, such as cervicofacial and abdominal diseases. This may be related to the greater incidence of disseminated disease in the thoracic form and may also be a reflection of late diagnosis in this condition. However, when the infection is recognized early and antibiotic treatment is given, the condition has an excellent prognosis with very low mortality as was in our case. Every physician should be familiar with this important differential in any patient with long-standing pulmonary infiltrates to prevent unnecessary morbidity or even unwarranted surgery.[6] It is, especially, important to consider this diagnosis where a soft-tissue mass is associated with an underlying consolidation. Surgical excision is not warranted as pulmonary actinomycosis responds adequately to conservative management with prolonged antibiotics.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Rippon JW. Medical Mycology. In: Wonsiewicz MJ, editor. Medical Mycology: The Pathogenic Fungi and the Pathogenic Actinomycetes. 3rd ed. Philadelphia: W.B. Saunders Co; 1988. p. 30-52.  Back to cited text no. 1
    
2.
Bennhoff DF. Actinomycosis: Diagnostic and therapeutic considerations and a review of 32 cases. Laryngoscope 1984;94:1198-217.  Back to cited text no. 2
    
3.
Apothéloz C, Regamey C. Disseminated infection due to Actinomyces meyeri: Case report and review. Clin Infect Dis 1996;22:621-5.  Back to cited text no. 3
    
4.
Russo TA. Agents of actinomycosis. In: Mandell GL, editor. Principles and Practice of Infectious Disease. 5th ed. New York: Churchill Livingstone; 1995. p. 2645-54.  Back to cited text no. 4
    
5.
Weese WC, Smith IM. A study of 57 cases of actinomycosis over a 36-year period. A diagnostic 'failure' with good prognosis after treatment. Arch Intern Med 1975;135:1562-8.  Back to cited text no. 5
    
6.
Mabeza GF, Macfarlane J. Pulmonary actinomycosis. Eur Respir J 2003;21:545-51.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2]



 

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