Surfactant for a Patient with Refractory Pyopneumothorax and Acute Respiratory Distress Syndrome Due to Pneumococcal Necrotizing Pneumonia Complicated by a Bronchopleural Fistula

Introduction

Necrotizing pneumonia is a rare complication of pneumonia in children, but its incidence is increasing.^1,2 Importantly, it can be associated with significant morbidity and mortality. ³ If not treated adequately it can lead to such complications as bronchopleural fistula, empyema, and sepsis. ¹ Staphylococcus aureus is the most common causative agent, followed by Streptococcus pneumoniae. Although surgical drainage is necessary in some cases, antimicrobial therapy is the cornerstone of management. ² Ideally, surgical interventions are kept to a minimum, but this is not always possible if there is a mass effect from air and fluid in the pleural space, pulmonary necrosis leading to massive hemoptysis, uncontrolled sepsis, or difficulties with assisted ventilation. ² Acute respiratory distress syndrome (ARDS) is also associated with necrotizing pneumonia. ³

Corticosteroids, inhaled nitric oxide (iNO), exogenous surfactant, and neuromuscular blocking drugs are some of the pharmacological treatments used in pediatric ARDS patients. ⁴ Surfactants are most commonly used to treat neonatal respiratory distress syndrome; however, surfactant dysfunction can occur in children and adolescents with ARDS, and exogenous surfactant use can be considered in such patients, although its use is controversial. ⁵ Although some pediatric ARDS studies show that surfactant replacement therapy decreases the need for mechanical ventilation and the mortality rate, and improves oxygenation and ventilation, the findings are far from being conclusive.^5,6

Moreover, there is a lack of consensus regarding surfactant administration procedures, such as through an endotracheal tube under invasive mechanical ventilation, through a thin endotracheal catheter under noninvasive ventilation, bronchoscopic administration, and nebulization.^5,6 The present report aimed to describe a patient with pediatric ARDS associated with necrotizing pneumonia and successfully treated with surfactant, so as to increase clinician knowledge of surfactant use in pediatric ARDS patients.

Case Report

A female patient aged 3 years and 7 months—the first child of consanguineous parents—presented to our hospital with a 5-day history of cough and new onset rapid breathing. The patient and family medical histories were unremarkable. All vaccines, including 4 doses of pneumococcal conjugate vaccine at age 2, 4, 6, and 12 months, were administered according to Turkey's national immunization program. Upon physical examination there was tachypnea (56 min⁻¹), tachycardia (160 min⁻¹), and no respiratory sounds at the base of the right hemithorax. In addition, bilateral rales, subcostal and intercostal retractions, and a Bacillus Calmette-Guérin scar on the upper left arm were noted.

Laboratory assessment showed an elevated white blood cell count (19,870 mm⁻³) with 82% neutrophils, an elevated C-reactive protein level (285 mg/L), and a low albumin level (2.3 g/dL). Liver and kidney function tests were normal. The initial arterial blood gas analysis while breathing through a non-rebreather reservoir bag oxygen mask showed respiratory acidosis with a pH of 7.19 and partial CO₂ pressure of 55 mmHg. Oxygen saturation was 93%. Chest roentgenogram showed pleural effusion on the right side. Axial chest computed tomography (CT) showed large right pleural effusion, with right lower and middle lobe collapse. Nasopharyngeal swab tests for reverse transcription-polymerized chain reaction (RT-PCR) for SARS-CoV-2 and blood culture were negative.

The patient was hospitalized, and ceftriaxone and vancomycin were initiated. A chest tube was inserted on the right side. The drained pleural fluid was exudate, and Gram-positive diplococci were observed through microscopic examination. S. pneumoniae grew in the culture. Acid-fast bacilli stain, PCR test, and culture were negative for Mycobacterium tuberculosis complex. Pleural effusion was not observed through thoracic ultrasonography performed after chest tube insertion. Immunological evaluation, including the absolute neutrophil and lymphocyte counts, serum immunoglobulins (IgG, IgA, IgM, IgE, and IgG subtypes), isohemagglutinin titer, peripheral lymphocyte subsets, activation response to phytohemagglutinin and anti-CD3, recent thymic emigrant CD4+ T cells ratio, dihydrorhodamine test, and total complement activity (CH50) were normal.

On day 3 of hospitalization sudden hypoxia developed, and respiratory sounds could not be heard in the left lung. The patient was quickly intubated. Chest X-ray showed bilateral pneumothorax, and a chest tube was inserted on the left side and a second tube was inserted on the right side. A second tube was inserted on the left side 2 days later, as the pneumothorax could not be controlled. Despite the chest tube insertions, bilateral pneumothorax persisted based on chest radiographs. Air leakage was measured between 70% and 90% in the mechanical ventilator; therefore, a pressure-controlled mode was preferred.

A follow-up chest CT on day 15 of hospitalization showed a septated pneumothorax and multiple cavitations in the right lung, and diffuse consolidation in the left lung (Fig. 1). A bronchopleural fistula was observed between the posterior segmental bronchus of the right lower lobe and the posterobasal pleural space (Fig. 1). These findings were interpreted as necrotizing pneumonia accompanied by a bronchopleural fistula. Because of the need for high positive pressure ventilation and diffuse collapse of the lungs, in addition to the antimicrobial treatment she was receiving, neuromuscular blockage was added to the treatment regimen and intratracheal bovine surfactant (beractant) was administered in 3 doses at 12-h intervals (total: 100 mg/kg) through the endotracheal tube blindly, based on the indication of pediatric ARDS, on day 16 of hospitalization.

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FIG. 1.

Chest CT. Septated pneumothorax and multiple cavitations in the right lung, and diffuse consolidation in the left lung. There is a bronchopleural fistula between the posterior segmental bronchus of the right lower lobe and the posterobasal pleural space (arrows). CT, computed tomography.

Subsequent radiographs showed that aeration in both lungs improved, although improvement was greater in the left lung (Fig. 2). Owing to the improved lung compliance, driving pressure was decreased from 16 to 14 cm H₂O, positive end-expiratory pressure was decreased from 12 to 9 cm H₂O, and the oxygenation index was decreased from 19.7 to 16 a few hours after surfactant treatment was completed. Then, on a daily basis the support provided by the ventilator was reduced. Air leakage measured in the ventilator decreased to 20%–30%, and she was extubated on day 28 of hospitalization. Air leakage from the chest tube decreased day by day and disappeared. The bronchopleural fistula healed without surgical repair, antibiotics were given for 6 weeks, and the patient was discharged on day 43 of hospitalization without any sequelae.

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FIG. 2.

Radiographs obtained before (a) and after (b) the administration of surfactant. Improved bilateral aeration, particularly in the nondiseased left lung after exogenous surfactant therapy.

Discussion

Herein we presented a patient with necrotizing pneumonia complicated by a bronchopleural fistula, who underwent intercostal drainage for pyopneumothorax, long-term mechanical ventilation, and was hospitalized for 43 days, and was then discharged without sequelae. An Indian study that evaluated 272 children with pneumonia reported that necrotizing pneumonia was detected in 10 patients, intercostal drainage was administered in 70%, mechanical ventilation was required in 70%, the duration of drainage was 9 days, and the mean duration of hospitalization was 13.5 days. ⁷

The primary problem in the presented patient was necrotizing pneumonia complicated by a bronchopleural fistula and pneumothorax that could not be controlled despite insertion of 4 chest tubes. Bronchopleural fistula occurs when lung necrosis passes through the pleura, resulting in communication between the pleural space and the lung. Pneumonia complicated by a bronchopleural fistula is associated with an increase in morbidity, longer pleural drainage time, and longer hospitalization. ¹

A study that evaluated 1,295 children with community-acquired pneumonia reported that 47 of the children had necrotizing pneumonia, 36 underwent pulmonary resection, 3 were complicated by a postoperative bronchopleural fistula, and 2 died due to multiple organ failure. ⁸ Another study that included 20 pediatric patients with bronchopleural fistula reported that early surgical intervention with insertion of a serratus anterior muscle digitation flap was effective and safe, and prevented morbidity associated with conservative management and necrotic lung resection surgery. ⁹ Owing to pneumothorax with a high percentage of air leakage, bilateral diffuse collapse of the lungs, and insufficient oxygenation in the presented patient, surgical treatment is considered, but due to the fact that the patient could not tolerate the surgery because of hemodynamic reasons and the complications associated with the surgery, especially in the early period, medical treatment was determined to be the most appropriate option.

In pediatric ARDS patients, especially severe cases with difficult oxygenation and ventilation, pharmacological treatments, such as neuromuscular blockers, corticosteroids, iNO, and surfactant, and such nonpharmacological treatments as the prone position are options; however, when these are not sufficient, extracorporeal membrane oxygenation (ECMO) can be considered.^5,6 Clinicians should keep in mind that all these treatment methods are controversial. From among these treatment options, the presented patient received neuromuscular blocker and surfactant treatment. The prone position could not be used in the presented case due to mechanical ventilation and bilateral chest tubes placed anteriorly. Surgical treatment of the septated pneumothorax was considered, but ultimately not used due to the associated complications. In addition, throughout the treatment process the treatment team and equipment were kept ready for venovenous ECMO. The high rate of ECMO complications prompted us to delay this treatment as much as possible.

The presented case is considered novel due to her improvement in response to surfactant treatment. Mechanical ventilation management was challenging, and every other intervention, including surgery and ECMO, carried too much risk for the patient. Septal pneumothorax with a bronchopleural fistula further complicated the ventilation. Under these circumstances, increasing the ventilation of even a single alveolus was highly valuable, which led to the idea of using exogenous surfactant. The treatment was observed to improve compliance of the nondiseased portions of the lung and facilitated improved re-expansion and subsequent ventilation in these segments (Fig. 2).

Surfactant treatment is considered to be useful in mechanically ventilated children with secondary surfactant inactivation due to severe pneumonia, but overinflation increases risk of air leaks such as pneumothorax and pulmonary interstitial emphysema. ¹⁰ However, surfactant treatment was well tolerated by the presented patient.

To the best of our knowledge this report is the first to describe surfactant treatment in a pediatric patient with ARDS due to necrotizing pneumonia. Although there is a lack of consensus regarding the use of surfactants in pediatric ARDS patients, there is evidence that surfactant treatment improves oxygenation, the PaO₂:FiO₂ ratio, the oxygenation index, and pH in such patients.^11,12 Surfactants can be considered as a salvage treatment in patients in which surgery is contraindicated and in those in which pneumothorax caused by a bronchopleural fistula severely impairs oxygenation and ventilation, as in the presented patient.