Lemierre syndrome complicated by bronchopleural fistula

ABSTRACT

We present a 19-year-old woman, a case of Lemierre syndrome, who presented with fever, sore throat, and left shoulder pain. Imaging revealed a thrombus in the right internal jugular vein, multiple nodular shadows below both pleura with some cavitations, right lung necrotizing pneumonia, pyothorax, abscess in the infraspinatus muscle, and multiloculated fluid collections in the left hip joint. After inserting a chest tube and administering urokinase for the pyothorax, a bronchopleural fistula was suspected. The fistula was identified based on clinical symptoms and computed tomography scan findings. If a bronchopleural fistula is present, thoracic lavage should not be performed as it may cause complications such as contralateral pneumonia due to reflux.

Introduction

Lemierre syndrome (LS) is a rare clinical disease caused by an acute oropharyngeal infection with internal jugular vein thrombosis, forming septic emboli in different organs. It is referred to as the killer sore throat and was considered to be fatal in up to 90% of patients in the pre-antibiotic era.[1,2] Generally, the management of LS includes antibiotic therapy and surgical drainage of abscesses and empyema. The adverse effects of using antifibrinolytic agents for treating empyema include major bleeding, which is usually intrapleural, chest pain, fever, and rash; but compared to utilizing only a chest tube drainage (which has a failure rate of 27%), the use of fibrinolytics results in fewer overall treatment failures.[3] We report our experience in managing a young female patient with LS with secondary empyema who developed a bronchopleural fistula during thoracic lavage.

Case Report

A 19-year-old healthy Japanese woman presented to a nearby hospital due to left shoulder pain after experiencing upper respiratory symptoms. She did not have diabetes or immunosuppressive diseases, and she was a non-smoker and was not a daily alcoholic beverage drinker. She was referred to our hospital because her chest CT scan showed bilateral diffuse reticular shadows, and magnetic resonance imaging of the left shoulder revealed an abscess in the infraspinatus muscle. On examination, her Glasgow Coma Scale score was E4M5V6, and she had a high-grade fever (39.6°C), was tachycardic (heart rate, 130 beats per minute), hypotensive (blood pressure, 98/56 mm Hg), tachypneic (respiratory rate, 24 breaths per minute), and her quick Sequential Organ Failure Assessment score was 2. Her laboratory tests revealed a white blood cell count of 16,300 cells/μL, a neutrophil level of 86.0%, and an elevated C-reactive protein (CRP) level of 15.17 mg/dL [Figure 1]. To rule out a prothrombotic state, thrombo-coagulation markers were evaluated, and the results revealed no abnormality. Upon performing a physical examination, an egg-sized mass was detected on the right side of her neck, and pain in the left hip joint and left shoulder was observed upon movement. Contrast-enhanced neck, chest, and abdominal CT scans were performed [Figure 2]. Ultrasonography showed a complete thrombus in the right jugular vein, which was in front of the abscess, as confirmed by formal Doppler imaging [Figure 3]. On second day of hospitalization, the left infraspinatus abscess was drained and it grew F. nucleatum. Blood cultures showed no growth of microorganisms. For LS treatment, the patient received intravenous antibiotic therapy with sulbactam sodium with ampicillin sodium (SBT/ABPC). However, it was modified to meropenem hydrate due to persistent high CRP levels. Later on SBT/ABPC were restarted after confirming the drug sensitivity of F. nucleatum. Anticoagulation therapy with heparin infusion followed by rivaroxaban was then initiated on day 7. On day 4 and 7 of hospitalization, drainage of the left purulent hip arthritis and right thoracic empyema was performed, respectively. The initial drainage of thoracic empyema was satisfactory. Hence 500 mL of saline was flushed into the chest tube manually once a day to confirm the absence of an air leak. Two days later (on day 9), the amount of drainage decreased, and intrapleural injection of urokinase was administered.

Figure 1.

Progress after hospitalization. MEPM, meropenem hydrate; SBT/ABPC, sulbactam sodium/ampicillin sodium

Figure 2.

Coronal and axial views on computed tomography scans of the neck (a), chest (b and c), and abdomen (d) revealing abscesses (arrows). A thrombus is present in the right internal jugular vein; multiple nodular shadows below the bilateral pleura, with some cavitations and consolidative opacities in the right lower lobe of the lungs, are also seen; abscess in the infraspinatus muscle and left hip joint is also noted; right pyothorax with some air is observed

Figure 3.

Upon admission, a complete occlusion of the right internal jugular vein was observed on neck ultrasonography. Further, an abscess cavity was found on the anterior surface of the right internal jugular vein. CA, carotid artery; IJV, internal jugular vein

After administering urokinase into the thoracic cavity she started coughing and complained of tasting salty water. Therefore, the thoracic cavity lavage was discontinued. A bronchopleural fistula was suspected, and this was confirmed on a chest CT scan [Figure 4]. The thoracic drain tube was then placed until day 17, but drainage during that period was only 50 mL per day. Her symptoms improved gradually, and all her laboratory data returned to normal (white blood cell count of 6,800 cells/μL, and CRP level of <0.3 mg/dL). The thrombus in the right jugular vein reduced in size, but not completely. Additionally, the undrained cervical abscess partially persisted, whereas the infraspinatus abscess, drained purulent hip arthritis, and thoracic empyema improved. She was discharged with the bronchopleural fistula on day 21 [Figure 1] and was being monitored for a standby closure operation or endobronchial embolization in the future.[4,5]

Figure 4.

The presence of a bronchopleural fistula was confirmed on chest CT scan after drainage (arrows). Chest cavity and infiltrative shadow within the right lung parenchyma appeared to communicate with each other

Discussion

LS is a rare disease. As per definition, LS is an infectious condition characterized by serious systemic symptoms caused by an abscess in distant organs and thrombophlebitis of the internal jugular vein triggered by upper respiratory diseases. F. necrophorum or other F. species are the most common causative organisms.[6,7] In the present case, the presence of F. nucleatum was confirmed by culturing the left infraspinatus abscess. Upon imaging, multiple nodular shadows, cavitation, and pleural effusion were observed in both lung fields. F. necrophorum is an anaerobic bacterium, and drainage is indispensable. However, if a bronchopleural fistula is present, thoracic lavage should not be performed because of the possible backflow of drugs and bacteria into the bronchus and contralateral lung leading to a life threatening complication.

In the present case, when the extrapulmonary chest cavity was filled with empyema, the bronchopleural fistula was difficult to see on CT; however, after drainage, it was clearly depicted on CT. A bronchopleural fistula, which usually accompanies empyema, is a severe complication of lung cancer surgery or poorly controlled thoracic empyema.[8,9,10] In the present case, the bronchopleural fistula may have been formed because of the delay in starting drainage of the thoracic empyema, and we suspect that the bronchopleural fistula was covered with a blood clot or other substance before urokinase injection.

In cases where an air leak or bubble is evident on CT after drainage,[11] it can be immediately decided not to wash. However, it is undeniable that it is sometimes difficult to make such a judgment because of the possibility of gas production by bacteria or small amounts of air contamination due to procedures, such as pleurodesis.

Several reports about LS have been published. However, the present case is a rare case of bronchopleural fistula confirmed on CT imaging only after thoracic drainage. A careless intrapleural lavage with urokinase or other drugs carries the risk of contralateral pneumonia and hemorrhage. In the present case, anticoagulation therapy with heparin infusion followed by rivaroxaban had been initiated; which would have increased the risk of bleeding if thoracic lavage had been continued.[12,13] Our experience suggests that it would be prudent to do a CT chest scan before performing thoracic lavage in order to rule out presence of a bronchopleural fistula and further complications.

Conclusion

The present case of LS was complicated by development of a bronchopleural fistula. The bronchopleural fistula may have been formed because of the delay in starting drainage of the thoracic empyema. Before performing thoracic lavage a chest CT scan should be performed to rule out a bronchopleural fistula.

Declaration of patient consent

The authors certify that appropriate patient consent was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.