Utility of Endobronchial Valves in a Patient with Bronchopleural Fistula in the Setting of COVID-19 Infection: A Case Report and Brief Review

Nazanin Sheikhan, MD¹, Elizabeth J. Benge, MD¹, Amanpreet Kaur, MD¹, Jerome K Hruska, DO², Yi McWhorter DO³, Arnold Chung MD⁴

¹Department of Internal Medicine, HCA Healthcare; MountainView Hospital, Las Vegas, NV, USA

²Department of Pulmonology, HCA Healthcare; MountainView Hospital, Las Vegas, NV, USA

³Department of Anesthesiology Critical Care Medicine, HCA Healthcare; MountainView Hospital, Las Vegas, NV, USA

⁴MountainView Cardiovascular and Thoracic Surgery Associates, HCA Healthcare; MountainView Hospital, Las Vegas, NV, USA

Abstract

Patients with COVID-19 pneumonia frequently develop acute respiratory distress syndrome (ARDS), and in severe cases, require invasive mechanical ventilation. One complication that can develop in patients with ARDS who are mechanically ventilated is a bronchopleural fistula (BPF). Although rare, the frequency of BPF in patients with COVID-19 pneumonia is increasingly recognized. Here, we present a 48-year old man with BPF associated with COVID-19 pneumonia. Treatment with a commercial endobronchial valve (EBV) system resulted in reduced air leak allowing for tracheostomy placement. Our case adds to a growing body of evidence suggesting that the presence of COVID-19 pneumonia does not hinder the utility of EBV’s in the treatment of BPF’s.

Abbreviation List

• ARDS = acute respiratory distress syndrome
• BIPAP = Bilevel Positive Airway Pressure
• BPF = Bronchopleural Fistula
• COVID-19 = Coronavirus Disease-2019
• CT = Computed Tomography
• CTA = Computed Tomography Angiography
• EBV = Endobronchial Valve
• HFNC = High Flow Nasal Cannula
• ICU = Intensive Care Unit
• RML = Right Middle Lobe
• RUL = Right Upper Lobe
• SARS-CoV-2 = Severe Acute Respiratory Syndrome Coronavirus-2
• VATS = Video-Assisted Thoracoscopic Surgery

Introduction

The COVID-19 pandemic has resulted in over one hundred million infections worldwide, in addition to millions of deaths (1). A less common sequelae of COVID-19 is bronchopleural fistula (2). A bronchopleural fistula is an abnormal sinus tract that forms between the lobar, main stem, or segmental bronchus, and the pleural space (3). BPF is typically treated by surgical repair, via a video-assisted thoracoscopic surgical approach (VATS) (3). Bronchoscopic approach with placement of airway stents, coils or transcatheter occlusion devices can be considered for those who are not suitable for surgical intervention (3).  A newer therapeutic modality for bronchopleural fistulae are endobronchial valves, which have been used successfully to treat COVID-19 patients diagnosed concurrently with bronchopleural fistulae (4). 

Here, we present a case of a critically ill patient developing a bronchopleural fistula with a concurrent COVID-19 infection, whose respiratory status was stabilized with an endobronchial valve.  To our knowledge, this is one of four case reports of a bronchopleural fistula arising in the setting of COVID-19.

Brief Review of Endobronchial Valves in COVID-19

Several other studies report success using endobronchial valves to treat bronchopleural fistulae in patients with COVID-19 pneumonia. One case series documents two cases of COVID-19 pneumonia complicated by bacterial super-infections, in which both patients experienced pneumothorax and persistent air leaks after mechanical invasive ventilation.  Both patients were successfully treated via EBV positioning. These researchers speculate that the severe inflammation associated with COVID-19 related ARDS induces inflammatory-related tissue frailty, pre-disposing lung tissue to damage via barotrauma, and the subsequent development of BPF (5).  

Another case documents the treatment of a 49-year-old male with COVID-19 pneumonia who was treated with steroids and tocilizumab. He also had a 3-week history of persistent air leak, which was successfully treated with an EBV. This team emphasizes that the thick, copious sections evident in patients afflicted by COVID-19 pose a risk for EBV occlusion. They highlight the importance of medically optimizing the patient and draining the air leak to mitigate the potential of this procedural complication developing (4).

In conjunction with the treatment course presented in our case, these case reports provide compelling evidence indicating that endobronchial valves can be successfully used to treat persistent air leaks in patients with COVID-19 pneumonia.

Case Presentation

Our patient is a 48-year-old male with a medical history significant for essential hypertension and Type 1 diabetes mellitus who presented to the emergency department complaining of acute onset generalized weakness, shortness of breath, and a near-syncopal event that had occurred the day prior. Vital signs on admission showed an oxygen saturation of 86% on ambient air, respiratory rate of 18 breaths per min, heart rate of 111 beats per min with a temperature of 37.6°C. He was tested for SARS-CoV-2 on admission and was found to be positive.

Initial computed tomography (CT) chest showed diffuse bilateral ground-glass opacities compatible with COVID-19 pneumonia. On admission, his inflammatory markers were elevated, with C-reactive protein 4.48 mg/dL, ferritin 1230 ng/ml, lactate dehydrogenase 281 IU/L, and D-dimer 0.76 mg/L. He received 1 dose of tocilizumab, convalescent plasma, as well as 5-day course of Remdesivir. His oxygen requirement increased as well as his work of breathing requiring High Flow Nasal Cannula (HFNC) and subsequently Bilevel Positive Airway Pressure (BiPAP); patient was transferred to the medical intensive care unit (ICU) 17 days after admission requiring intubation. Computed tomography angiography (CTA) chest could not be obtained to rule out pulmonary embolism as patient was too unstable. Patient was started on Heparin drip empirically which had to be discontinued due to gastrointestinal bleeding. He had worsening oxygenation, ventilator asynchrony, with P:F ratio of 47, requiring high-dose sedation and neuromuscular blockade, as well as prone positioning. Repeat CT chest on day 21 demonstrated bilateral pneumothoraces and pneumomediastinum as well as interval worsening of diffuse ground glass infiltrates (Figure 1), requiring bilateral chest tube placement.

Figure 1. Computed tomography chest showing pneumomediastinum, bilateral pneumothoraces, and diffuse ground glass attenuation of the lungs bilaterally.

On the 34th day of admission, he developed a right-sided tension pneumothorax likely secondary to ongoing severe ARDS, requiring replacement of dislodged right chest tube. Patient subsequently had worsening of right pneumothorax requiring an additional second chest tube placement. Patient developed persistent air leak concerning for right bronchopleural fistula. On hospital day 42, patient underwent intrathoracic autologous blood patch with persistence of large air leak. After interdisciplinary conference with cardiothoracic surgery, pulmonary, and the ICU team, it was decided that patient is not a surgical candidate hence interventional pulmonology was consulted for EBV placement to facilitate chest tube removal and ventilator weaning.

Patient underwent fiberoptic bronchoscopy on hospital day 52; pulmonary balloon was used to sequentially block the right mainstem, bronchus intermedius, and basilar segments. The air leak was recognized to be coming from right middle lobe (RML) and the apex of the right upper lobe (RUL) status post placement of two endobronchial valves in the medial and lateral segments of the RML (Figure 2).

Figure 2. Bronchoscopic view of endobronchial valves.

The RUL could not be entered secondary to angulation and technical inability of the instruments to achieve a sharp bend. Post-bronchoscopy, patient had 50 mL reduction in air leak resulting in improvement of his ventilator settings such that a tracheostomy could be safely performed. Left-sided chest tube was removed with resolution of pneumothorax. Repeat CT chest on hospital day 115 demonstrated persistent right bronchopleural fistula (Figure 3).

Figure 3. Computed tomography chest showing bronchopleural fistula in the right middle lobe and collapsed and shrunken right middle lobe with endobronchial occlusion stents at the central airway. Yellow arrow showing endobronchial valves and red arrows showing bronchopleural fistula

The patient is currently pending transfer to a long-term acute care hospital for aggressive physical therapy and eventual transfer to a tertiary center for lung transplantation evaluation.

Discussion

Scientific research has moved at an unprecedented speed in an attempt to shed light on the manifestations of COVID-19. The most common presentation of COVID-19 includes cough, fever, shortness of breath, and new onset anosmia and ageusia (6).

Common complications include coagulopathy, pulmonary emboli, and in severe cases, acute respiratory distress syndrome (7). Bronchopleural fistulae have emerged as a rare but known complication of COVID-19. This pathology is traditionally seen as a post-surgical complication arising from lobectomy or pneumonectomy (8). All cause mortality secondary to bronchopleural fistulae are high; with mortality rates ranging from 18-67% (8).

A relatively novel therapeutic modality for bronchopleural fistulae are endobronchial valves, which have been used in patients who are not candidates for surgery, such as our patient (9). They work as a one-way valve that allow the pathologically trapped air to exit the respiratory system, but not enter (4).

Differential diagnoses for bronchopleural fistulae include alveolar pleural fistulas and empyema (11). Alveolar pleural fistulas are abnormal communications between the pulmonary parenchyma, distal to a segmental bronchus, and the pleural space, while bronchopleural fistulas are more proximal; representing abnormal connections between a mainstem, lobar, or segmental bronchus and the pleural space (12). These pathologies are differentiated with direct visualization on bronchoscopy, as was demonstrated in our patient (12).

There are currently no official statistics on the epidemiology of bronchopleural fistulae in COVID-19. A disappointing aspect of our case was the lack of complete resolution of the patient’s air leak after the placement of the endobronchial valve. While the patient’s condition did improve after the valve was placed, he continued to suffer from respiratory illness related to his bronchopleural fistula. Although complete remission was not achieved, the endobronchial valve placement did facilitate respiratory recovery sufficient enough to facilitate a tracheostomy. The patient was then stabilized for eventual transfer to a long-term acute care facility, where he will undergo physical therapy and await lung transplantation. It is important to emphasize that while the endobronchial valve was not curative, it stabilized the patient for possible future curative treatments.  

Conclusion

Despite their rarity, bronchopleural fistulas are a pulmonary complication of COVID-19. Although the insertion of the endobronchial valve in our patient resulted in a reduction of the air leak as opposed to complete resolution, this case still emphasizes a therapeutic benefit of endobronchial valves in such instances. Overall, our case demonstrates the importance of clinical vigilance in the face of unusual pulmonary complications related to COVID-19, and that treatment of these complications requires flexibility and creativity.

References

1. WHO Coronavirus (COVID-19) Dashboard [Internet]. World Health Organization. World Health Organization; [cited 2021May31]. Available from: https://covid19.who.int/ 
2. Hopkins C, Surda P, Kumar N. Presentation of new onset anosmia during the COVID-19 pandemic. Rhinology. 2020 Jun 1;58(3):295-298. [CrossRef] [PubMed]
3. Miesbach W, Makris M. COVID-19: Coagulopathy, Risk of Thrombosis, and the Rationale for Anticoagulation. Clin Appl Thromb Hemost. 2020 Jan-Dec;26:1076029620938149. [CrossRef] [PubMed]
4. Talon A, Arif MZ, Mohamed H, Khokar A, Saeed AI. Bronchopleural Fistula as a Complication in a COVID-19 Patient Managed With Endobronchial Valves. J Investig Med High Impact Case Rep. 2021 Jan-Dec;9:23247096211013215. [CrossRef] [PubMed]
5. Donatelli P, Trenatacosti F, Pellegrino MR, et al. Endobronchial valve positioning for alveolar-pleural fistula following ICU management complicating COVID-19 pneumonia. BMC Pulm Med. 2021 Sep 27;21(1):307. [CrossRef] [PubMed]
6. Salik I, Vashisht R, Abramowicz AE. Bronchopleural fistula. StatPearls [Internet]. 2020 Aug 27. [CrossRef]
7. Cardillo G, Carbone L, Carleo F, Galluccio G, Di Martino M, Giunti R, Lucantoni G, Battistoni P, Batzella S, Dello Iacono R, Petrella L, Dusmet M. The Rationale for Treatment of Postresectional Bronchopleural Fistula: Analysis of 52 Patients. Ann Thorac Surg. 2015 Jul;100(1):251-7. [CrossRef] [PubMed]
8. Sarkar P, Chandak T, Shah R, Talwar A. Diagnosis and management bronchopleural fistula. Indian J Chest Dis Allied Sci. 2010 Apr-Jun;52(2):97-104. [PubMed]
9. Pathak V, Waite J, Chalise SN. Use of endobronchial valve to treat COVID-19 adult respiratory distress syndrome-related alveolopleural fistula. Lung India. 2021 Mar;38(Supplement):S69-S71. [CrossRef] [PubMed]
10. Musani AI, Dutau H. Management of alveolar-pleural fistula: a complex medical and surgical problem. Chest. 2015 Mar;147(3):590-592. [CrossRef] [PubMed]
11. Mehta HJ, Malhotra P, Begnaud A, Penley AM, Jantz MA. Treatment of alveolar-pleural fistula with endobronchial application of synthetic hydrogel. Chest. 2015 Mar;147(3):695-699. [CrossRef]  [PubMed]

Acknowlegements

This research was supported (in whole or in part) by HCA Healthcare and/or an HCA Healthcare affiliated entity. The views expressed in this publication represent those of the author(s) and do not necessarily represent the official views of HCA Healthcare or any of its affiliated entities. 

Cite as: Sheikhan N, Benge EJ, Kaur A, Hruska JK, McWhorter Y, Chung A. Utility of Endobronchial Valves in a Patient with Bronchopleural Fistula in the Setting of COVID-19 Infection: A Case Report and Brief Review. Southwest J Pulm Crit Care. 2021;23(4):109-14. doi: https://doi.org/10.13175/swjpcc046-21 PDF 

A New Interventional Bronchoscopy Technique for the Treatment of Bronchopleural Fistula

Evan Denis Schmitz, MD

Abstract

A patient receiving mechanical ventilation with multiple left hydropneumothoraces had a persistent air leak through the thoracostomy tube. The leak was temporarily resolved by interventional bronchoscopy at the bedside in the ICU. Because of the limited resources available at the hospital, a Swan-Ganz catheter was inserted into the left upper lobe bronchus, inflated and left in place. The air leak ceased and the left upper lobe bronchus was occluded with an autologous blood plug by infusing the patient’s own blood through the distal port of the catheter. The patient’s oxygenation improved significantly. The effects persisted for 2.5 hours until the air leak returned while the patient remained intubated. Such a technique may be useful when managing persistent air leaks.

Introduction

An air leak during mechanical ventilation despite the insertion of a thoracostomy tube can be detected by the bubbling of air through the air seal in the chest drainage system (1). A persistent air leak (PAL) is often defined as persistence of the air leak beyond 24 hours, which can hinder ventilation and inhibit lung expansion. Furthermore, the leak may inhibit healing of the fistula between the lung and the pleural space. Recommendations for the management of PALs include surgical repair as the gold standard for treatment (1,2). However, published anecdotal reports describe successful treatment of PALs with endobronchial insertion of fibrin sealants, ethanol injection, metal coils, Watanabe spigots and endobronchial valves. Success is also reported with chemical and autologous blood patch pleurodesis (1). We report a bedside interventional bronchoscopy technique using a Swan-Ganz catheter for the treatment of PALs while intubated and ventilated. A Swan-Ganz catheter is inserted into a lobar bronchus using direct visualization with a bronchoscope, the balloon is inflated and left in place while an autologous blood plug is created utilizing the distal port of the catheter.

Case Presentation

A 69-year-old man with no prior medical contact presented to the emergency department with severe shortness of breath and altered mental status. He was intubated in the emergency department for hypoxia. On arrival to the ICU he was in hypoxic respiratory failure and septic shock with a PaO₂ in the 40s. His ventilator plateau pressures were 40-50 cm H₂O. Chest radiography revealed moderate pneumomediastinum and multiple loculated hydropneumothoraces involving the left lung with suspected necrotic left upper lobe (Figure 1).

Figure 1. Portable AP chest x-ray showing left lung loculated hydropneumothoraces in the apex, medial and lateral walls of the left chest, subcutaneous emphysema, mediastinal emphysema and very low lung volumes. There are right apical and lower lobe areas of consolidation. A left thoracostomy tube is in place.

A 32 Fr thoracostomy tube was placed in the left intercostal space lateral to the nipple in the mid-axillary. The larger thoracostomy tube was chosen because of concern that the smaller pig-tailed catheters might not be adequate to control the leak. Plateau pressure improved to 30 cm H2O.

Despite a low tidal volume ventilator strategy and -40 cm H₂O suction through the thoracostomy tube, the patient had an air leak through the thoracostomy tube which continued to bubble in the water seal chamber during both inspiration and expiration. The air leak did not improve over the ensuing 24 hours and subcutaneous emphysema worsened when attempts were made to decrease suction which was confirmed by physical exam and chest x-ray. Selective right lung ventilation led to inadequate ventilation as evidenced by increasing end-tidal CO_2.

To determine and attempt to control the source of the persistent air leak, an interventional bronchoscopy was performed at bedside. Because other devices to such as metal coils, endobronchial valves, fibrin glue and a YAG laser were unavailable, a 6 Fr Swan-Ganz catheter was used. The Swan-Ganz catheter was threaded through the opening of the bronchoscope adaptor down the endotracheal tube to 3 cm above the carina. A flexible bronchoscope was then advanced along the side of the catheter through the bronchoscope adaptor and down the endotracheal tube. The catheter was not inside the working channel of the bronchoscope. The catheter was manipulated along with the bronchoscope, taking advantage of the inherent bend in the catheter, into the left mainstem bronchus and into the left upper lobe bronchus just distal to the lingular bronchus and inflated (Figure 2).

Figure 2. Panel A: Bronchoscopic view showing the Swan-Ganz catheter in the left upper lobe bronchus. Panel B: Chest x-ray confirming the Swan-Ganz catheter in the left upper lobe with the balloon inflated (arrow).

The massive air leak stopped completely. A blood plug was then created by instilling 20 ml of the patient’s own blood into the distal port of the catheter distal to the balloon along with 5 ml of 1:1000 epinephrine. The bronchoscope was used to hold the balloon in place for 10 minutes while the blood clotted. The bronchoscope was carefully removed and the catheter with the balloon inflated was left in place (Figure 3).

Figure 3. Bronchoscopic view showing the catheter passing into the left upper lobe bronchus with the surrounding blood plug.

The bronchoscope adaptor was taped post-bronchoscopy at the opening with an occlusive dressing so no air could leak around the catheter. The patient tolerated the procedure well. The air leak was successfully stopped with no evidence of worsening pneumothoraces. After PaO₂ increased from the 40s on admission to the 170s after the PAL was stopped. Chest x-ray at 1 and 3 hours showed no evidence of worsening pneumothorax with the Swan-Ganz catheter still in place and inflated in the left upper lobe bronchus. After 2.5 hours, a smaller air leak did return but was present only during inspiration.

Discussion

A PAL during mechanical ventilation can be a serious complication of ventilator therapy. It can lead to poor lung expansion, ventilation/perfusion mismatch, direct extension of airway infection into the pleural space, and an inability to maintain positive end-expiratory pressure. Patients with a PAL have increased complications, including ICU readmission, pneumonia, and a longer hospital stay (3,4). Fortunately, it appears to be relatively rare. In a retrospective study only 39 out of 1,700 mechanically ventilated patients had a PAL defined as lasting for greater than 24 hours (5).

The American College of Chest Physicians guidelines published in 2001 and the 2010 British Thoracic Society guidelines on pleural disease recommend waiting for about 4 days and then seeking surgical evaluation for a PAL (2,6). It was recommended that consideration should be given to placing the thoracostomy tube to water seal rather than to suction. However, this may not be possible in patients with a large persistent air leak that complicates ventilation. In those instances, a variety of endobronchial and pleural interventions have been attempted. Although the reports are anecdotal, most achieved success with either none or minimal complications (1). There have been two basic approaches to treat PALs; sealing the air leak from the bronchial side or from the pleural side. Those therapies administered through the bronchoscope include fibrin sealant, metal coils, Watanabe spigots, synthetic hydrogel, platelet gel, endobronchial valves and YAG laser (1). Complications were infrequent and minor. Ethanolamine and ethanol have also been used but there appear to be more complications with those treatments. From the pleural side, blood patch and chemical pleurodesis have been used successfully (1). However, chemical pleurodesis might result in a trapped lung.

The technique reported here can be performed with materials available in the ICU. A torqueable guidewire can be inserted if needed to help increase the catheter stiffness and help with advancement of the catheter into the individual bronchus to identify the source of the bronchopleural fistula. Alternatives to a blood patch might include occlusion of the culprit bronchus with the patient’s own mucus and argon plasma coagulation to form a clot. A blood patch can be used to determine the potential success of a more permanent material to occlude the bronchus, such as a fibrin seal, synthetic hydrogel, laser, or before attempting endobronchial valve placement.

Conclusion

Bedside endobronchial management of PAL is feasible using a flexible bronchoscope and Swan-Ganz catheter for localization, tamponade and delivery of a blood plug.

References

1. Dugan KC, Laxmanan B, Murgu S, Hogarth DK. Management of persistent air leaks. Chest. 2017;152(2):417-23. [CrossRef] [PubMed]
2. Baumann MH, Strange C, Heffner JE. Management of spontaneous pneumothorax: an American College of Chest Physicians Delphi consensus statement. Chest. 2001;119(2):590-602. [CrossRef] [PubMed]
3. Liberman M, Muzikansky A, Wright CD, et al. Incidence and risk factors of persistent air leak after major pulmonary resection and use of chemical pleurodesis. Ann Thorac Surg. 2010;89(3):891-897. [CrossRef] [PubMed]
4. DeCamp MM, Blackstone EH, Naunheim KS, et al. Patient and surgical factors influencing air leak after lung volume reduction surgery: lessons learned from the National Emphysema Treatment Trial. Ann Thorac Surg. 2006;82(1):197-206. [CrossRef] [PubMed]
5. Pierson DJ, Horton CA, Bates PW. Persistent bronchopleural air leak during mechanical ventilation. A review of 39 cases. Chest. 1986;90(3):321-3. [CrossRef] [PubMed]
6. Havelock T, Teoh R, Laws D, et al. Pleural procedures and thoracic ultrasound: British Thoracic Society Pleural Disease Guideline 2010. Thorax. 2010;65(suppl 2):ii61-ii76. [CrossRef] [PubMed]

Cite as: Schmitz ED. A new interventional bronchoscopy technique for the treatment of bronchopleural fistula. Southwest J Pulm Crit Care. 2017;15(4):174-8. doi: https://doi.org/10.13175/swjpcc120-17 PDF 

February 2013 Critical Care Case of the Month: Thoracentesis Through the Looking Glass

Clement U. Singarajah MD

Jay E. Blum

Allen R. Thomas MD

Henry Luedy MD

Elijah Poulos MD

Tonya Whiting DO

Phoenix VA Medical Center

Phoenix, AZ

History of Present Illness

A 62 year old man was recently diagnosed with Stage 4 squamous cell left lung cancer with metastases to the pleura, brain and mediastinum. He also had known chronic obstructive pulmonary disease (COPD) with a FEV1 = 1.96 L and a known left side pleural effusion (see Figure 1).

Figure 1. Baseline chest radiograph showing left pleural effusion (red arrow).

He was seen as an outpatient for symptomatic shortness of breath and underwent real time ultrasound guided left sided thoracentesis removing 500 ml of straw-colored fluid. The procedure was uneventful except that near the end, the patient started to cough.  He denied any symptoms post procedure apart from some minor puncture site pain. A routine post procedure chest x-ray was performed (Figure 2).

Figure 2. Post-thoracentesis x-ray (Panel A) and its negative image (Panel B).

What new abnormality is identified on the post-procedure chest x-ray?

1. Left pneumothorax
2. Right pneumothorax
3. Lung “sliding” on the left
4. New pneumonia in the left upper lobe
5. Left hilar retraction

Reference as: Singarajah CU, Blum JE, Thomas AR, Luedy H, Poulos E, Whiting T. February 2013 critical care case of the month: thoracentesis through the looking glass. Southwest J Pulm Crit Care. 2013;6(2):63-74. PDF