Bronchopleural fistula (BPF) with empyema caused by severe necrotizing pulmonary infection is a complicated clinical problem that is often associated with poor general condition so surgical interventions cannot be tolerated in most cases. Here, we present the successful management of multiple BPF with empyema in a mechanically ventilated patient with aspiration lung abscess. Occlusion utilizing Gelfoam followed by endobronchial valves (EBVs) implanted inverted via bronchoscope decreased the air leaking significantly and made intrapleural irrigation for empyema achievable and safe. This is the first report of a novel way of EBV placement and the combination use with other occlusive substances in BPF with empyema in a patient on mechanical ventilation. This method may be an option for refractory BPF cases with pleural infection.

Established Facts

  • The repair of multiple bronchopleural fistulas with empyema under positive pressure ventilation is difficult.

  • Bronchoscopic intervention is an optional approach in the closure of bronchopleural fistula and various occlusive substances have been invented and successfully used so far.

Novel Insights

  • Inverted placement of one-way endobronchial valves with Gelfoam is a novel and effective method for complicated and refractory bronchopleural fistula cases with pleural infection.

Bronchopleural fistula (BPF), usually caused by surgical lung resection, is an intractable clinical problem with a poor prognosis. Less frequently, BPF can occur spontaneously in patients with necrotizing pneumonia and sometimes even complicated with empyema. In this setting, the poor vascularized tissue and nutrition status due to the severe infection will make fistula repair more challenging. Furthermore, if mechanical ventilation is initiated for respiratory failure, the situation will be more complicated. Herein, we describe a case of refractory multiple BPF and empyema under positive pressure ventilation in a patient with aspiration lung abscess who was treated by inverted endobronchial valve (EBV) placement and Gelfoam successfully.

A 49-year-old woman with a history of alcoholism and aspiration was admitted to the RICU with continuous fever, productive cough, and progressive dyspnea in recent 10 days. On admission, the patient’s temperature was 39°C accompanied by severe orthopnea and her PaO2 in blood gas was 59.3 mm Hg (face mask, O2 10 L/min). Chest X-ray showed lung abscess and hydropneumothorax located on the right side and bilateral infiltration. Blood tests revealed sepsis with multiple organ dysfunction including mild to moderate abnormalities of the liver, cardiac, and coagulation systems. She was intubated due to severe respiratory failure, and two chest tubes were placed in the fourth and ninth intercostal on the anterior and posterior axillary line, respectively, for the drainage of both air and fluid after admission. Intravenous antibiotics including meropenem and vancomycin, prophylactic anticoagulation, and comprehensive supporting treatment were also carefully prescribed. Prevotella intermedia and Pyramidobacter piscolens which are usually found in the mouth and gastrointestinal tract were detected in the microbial culture of sputum and pleural fluid. Persistent air leak and pus-like pleural fluid suggested the existence of BPF and empyema which were also confirmed by bronchoscope and computed tomography scan (shown in Fig. 1). During pleural irrigation with saline, large amounts of purulent fluid spilled from the right bronchus to the left were observed. To perform pleural irrigation effectively and avoid infection spreading, fistula closure would be the key solution for this patient. Since surgical procedures could not be tolerated for her poor general condition and local access to materials situation as well, we planned to insert EBV inverted combined with Gelfoam. For the inverted EBV loading, we used a needle with a cord to thread through the valve protector around the valve, threaded the cord into the compression region of funnel to pull the EBV into the compression region carefully until the EBV was completely within the region, and then removed the cord (shown in Fig. 2). The following preparation was the same as usual. B3a, B4, B8, B9, and B10 were identified as responsible bronchus by intrapleural instillation of methylene blue dilution and balloon test. After Gelfoam placement in each segment, two 4.0 EBV and one 5.0 EBV were inserted into B3a, B4, and B8+9+10, respectively (shown in Fig. 1). Air leakage decreased obviously, and no spilled methylene blue dilution was observed. Infection was controlled properly, and the oxygenation index was well improved in the following 2 weeks so that the patient was extubated successfully. On day 24 after the procedure, no air leaks were observed, and computed tomography scan showed that the right lung lobe was fully re-expanded (shown in Fig. 1); both chest tubes were removed. The EBVs were also removed on day 43 (shown in Fig. 1), and the patient was discharged with no respiratory support finally. The whole treatment process was shown in Figure 3. No recurrent fistula or pneumothorax was found in the follow-up visit.

Fig. 1.

a-c Inverted placement of EBVs following Gelfoam insertion in B3a, B4, and B8+9+10, respectively. d-f The bronchoscopic view after EBVs were removed. g-i The CT scan showed BPF on the right side caused by severe necrotizing pneumonia on admission. j-l BPF were closed and empyema absorbed shown in CT scan after the treatment. EBV, endobronchial valve; CT, computed tomography; BPF, bronchopleural fistula.

Fig. 1.

a-c Inverted placement of EBVs following Gelfoam insertion in B3a, B4, and B8+9+10, respectively. d-f The bronchoscopic view after EBVs were removed. g-i The CT scan showed BPF on the right side caused by severe necrotizing pneumonia on admission. j-l BPF were closed and empyema absorbed shown in CT scan after the treatment. EBV, endobronchial valve; CT, computed tomography; BPF, bronchopleural fistula.

Close modal
Fig. 2.

a A cord was threaded through the valve protector by a needle. b EBV was pulled into the compression region of the funnel with the cord. c EBV was completely within the compression region, and the cord was removed. EBV, endobronchial valve.

Fig. 2.

a A cord was threaded through the valve protector by a needle. b EBV was pulled into the compression region of the funnel with the cord. c EBV was completely within the compression region, and the cord was removed. EBV, endobronchial valve.

Close modal
Fig. 3.

The treatment process and outcome of the patient.

Fig. 3.

The treatment process and outcome of the patient.

Close modal

BPF is commonly encountered after lung resection, while necrotizing lung infections can sometimes also lead to refractory BPF [1]. No standardized recommendations or guidelines on BPF treatment have been published so far. Usually, apart from thoracic drainage, antibiotics, and intrapleural agents, bronchoscopic interventions and surgical repair are the main approaches for the treatment, and sometimes they can be complementary. For patients with a high risk for surgery or who need a bridge to surgery, bronchoscopic interventions should be considered. Previous studies showed that the success rate of BPF treated by bronchoscopic techniques ranged from 40% to 100% [2].

Here, we report a rapidly progressive aspiration lung abscess case leading to intubation and multiple organ dysfunction. Empyema and multiple BPFs were found due to severe lung tissue necrosis. For consideration of her unstable illness and diffused distributed fistulas, surgical repair was not suggested at that time. In addition to adequate systemic support and antibiotic therapy, the effective control of local infection in the pleural cavity through ongoing pleural drainage and intrapleural irrigation is also regarded as a requirement [3, 4]. However, pus-like fluid was found regurgitated into the bronchi, even the contralateral bronchi and lung since the existence of fistulas. Besides, continuous positive pressure ventilation made the situation more complicated in that positive end-expiratory pressure might be lost because of the fistulas, resulting in hypoxemia, decrease of tidal volume, and worsening of ventilation-perfusion mismatch. The addition of intermittent chest tube suction also increased flow across the fistula, leading to poor gas exchange and BPF healing [5]. Although the conventional mode of ventilation had been used, the fistulas were not improved effectively.

Independent lung isolation and ventilation could not be tolerated for her poor oxygenation, so we planned endobronchial intervention to close these fistulas. There are various occlusive substances including fibrin glue, tissue adhesive, Gelfoam, silver nitrate, sclerosants, spigot, EBV, stent, vascular coil, Amplatzer device, and autologous blood patch to choose from. EBV is originally designed for endoscopic lung volume reduction and has also been used for BPF with good tolerance and efficacy [6]. Recently, mesenchymal stem cells, fibroblast growth factor, and autologous fat graft were also reported as safe and effective methods for the closure of BPF [7‒9]. The size, number, location, and cause of the fistula, underlying disease status, patient’s condition, access to materials, and economic cost should be comprehensively considered in treatment and material planning. For this patient, the BPFs caused by necrotizing pneumonia were located in distal lung tissue which could not be observed in bronchoscopy, and thus tissue adhesive or sclerosants were not that suitable. The fistulas were small and diffused distributed so that stent or Amplatzer device was not considered as well. Occlusive substances were the main choices. However, commonly used materials alone could not be fixed to the responsible bronchi securely and tightly in this ventilated patient with empyema and pneumothorax, so we chose a combined way to occlude for a definite and effective closure. Gelfoam is cheap and readily available in our area, and inverted EBV placement could help to hold the Gelfoam in place and avoid possible dislodgement caused by fluid reflux from the pleural space so that thoracic irrigation could be feasible to achieve the control of empyema and infection could be stopped from spreading to the other side which was a critical problem for this patient at that time. If EBVs were inserted in a conventional way, the Gelfoam might not be fixed so well and EBVs also had risk of migration. Once the dislodgement happened or the Gelfoam was absorbed, the fluid in the pleural cavity might spill back again if the patient had not recovered from BPF. To some extent, inverted EBV might decrease the air leaking since the diameter of the valve is smaller than that of the retainer part, while air leaking could not be stopped totally. Gelfoam in the distal segmental bronchus could also help to seal the leaking completely. Besides, EBV is easy to remove and Gelfoam is absorbable if the patient recovered from BPF in a short time. Finally, air leaking was successfully stopped, and local irrigation was achieved to prevent intrapleural organization. It is regrettable that the endobronchial Watanabe spigot has not been approved in our area, which might be an alternative for this patient. Since the Gelfoam might be absorbed in 2–3 weeks and the inverted EBV could not stop potential air leaking to the pleural space under mechanical ventilation, the patient might develop chronic BPF. Therefore, we also had planned to remove the inverted EBV after the empyema was well solved and then place EBVs in a conventional way. Fortunately, this situation did not occur, and the fistulas healed completely.

We report a successful bronchoscopic intervention in the closure of multiple BPFs and empyema caused by necrotizing lung inspiration infections in a critically intubated patient on mechanical ventilation. Inverted placement of one-way EBVs with Gelfoam is a novel and effective method for complicated BPF cases with pleural infection. The combination and flexible use of available substances tailored to individual illnesses is a challenge for interventional pulmonologists. We also look forward to developing more effective and personalized devices or materials in this field.

We sincerely thank all the clinical staff who provided care for this patient in ICU and emergency room.

The research was conducted ethically in accordance with the World Medical Association Declaration of Helsinki. Written informed consent was obtained from the patient for publication of this case report and any accompanying images.

The authors have no conflicts of interest to declare.

This study was supported by grants from the National Key R&D Plan of China (2023YFB3506903), the Clinical Research Plan of SHDC (SHDC2020CR5010-002), the Shanghai Municipal Key Clinical Specialty (shslczdzk02201), and the Shanghai Joint Research Project of Emerging Frontier Technology (SHDC12022109).

Jie Liu wrote the original draft of the manuscript. Maosong Ye, Jie Liu, and Chun Li designed and performed the bronchoscopy interventions. Ling Ye and Qinjun Shen took care of the patient. Zilong Liu obtained the images through the clinical practice. Maosong Ye, Qunying Hong, and Yuanlin Song reviewed the manuscript. All authors approved the final draft of the manuscript for publication.

All data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.

1.
Ding
M
,
Gao
YD
,
Zeng
XT
,
Guo
Y
,
Yang
J
.
Endobronchial one-way valves for treatment of persistent air leaks: a systematic review
.
Respir Res
.
2017
;
18
(
1
):
186
. .
2.
Jin
L
,
Li
Y
.
Bronchoscopic interventions for bronchopleural fistulas
.
Ther Adv Respir Dis
.
2023
;
17
:
17534666231164541
. .
3.
Shen
KR
,
Bribriesco
A
,
Crabtree
T
,
Denlinger
C
,
Eby
J
,
Eiken
P
, et al
.
The American Association for Thoracic Surgery consensus guidelines for the management of empyema
.
J Thorac Cardiovasc Surg
.
2017
;
153
(
6
):
e129
46
. .
4.
Hooper
CE
,
Edey
AJ
,
Wallis
A
,
Clive
AO
,
Morley
A
,
White
P
, et al
.
Pleural irrigation trial (PIT): a randomised controlled trial of pleural irrigation with normal saline versus standard care in patients with pleural infection
.
Eur Respir J
.
2015
;
46
(
2
):
456
63
. .
5.
Grotberg
JC
,
Hyzy
RC
,
De Cardenas
J
,
Co
IN
.
Bronchopleural fistula in the mechanically ventilated patient: a concise review
.
Crit Care Med
.
2021
;
49
(
2
):
292
301
. .
6.
Giddings
O
,
Kuhn
J
,
Akulian
J
.
Endobronchial valve placement for the treatment of bronchopleural fistula: a review of the current literature
.
Curr Opin Pulm Med
.
2014
;
20
(
4
):
347
51
. .
7.
Zeng
Y
,
Gao
HZ
,
Zhang
XB
,
Lin
HH
.
Closure of bronchopleural fistula with mesenchymal stem cells: case report and brief literature review
.
Respiration
.
2019
;
97
(
3
):
273
6
. .
8.
Marchioni
A
,
Mattioli
F
,
Tonelli
R
,
Andreani
A
,
Cappiello
GF
,
Serafini
E
, et al
.
Endoscopic bronchopleural fistula repair using autologous fat graft
.
Ann Thorac Surg
.
2022
;
114
(
5
):
e393
6
. .
9.
Guo
S
,
Bai
Y
,
Li
Y
,
Chen
T
.
A large central bronchopleural fistula closed by bronchoscopic administration of recombinant bovine basic fibroblast growth factor: a case report
.
Respiration
.
2021
;
100
(
10
):
1000
4
. .