Treatment of intractable bronchopleural fistula with a one-side-closed silicone stent using retrograde approach: A case report

Abstract

A male patient (age: 85 -years) with lung cancer underwent basal segmentectomy. Subsequently, he underwent emergency open window thoracotomy for a bronchial stump fistula. The general and nutritional conditions of the patient improved; nevertheless, natural closure of the fistula did not occur. Therefore, the patient underwent fistula closure using an endobronchial Watanabe spigot, polyglycolic acid sheet and N-butyl-2-cyanoacrylate. Nine months later, the fistula had enlarged. The air leak was treated by applying pressure with gauze; however, this approach was not sufficiently effective, and the patient became unable to expectorate phlegm or speak. One side of a straight-type silicone stent with an outer diameter measuring 9mm was closed, and the stent was inserted into the fistula through the fenestration with the closed side at the tip. The L-sized endobronchial Watanabe spigot was placed into the lumen of the stent to reinforce it. Air leak from the fistula was significantly reduced, making breathing and expectoration easier. Retrograde closure of a bronchial fistula using a unilaterally closed silicon stent can be an effective treatment for large, refractory bronchial fistulas.

1. Introduction

Broncho pleural fistula (BPF) after pulmonary resection is a potentially fatal complication that adversely affects respiratory status. Although BPF after lobectomy is rare (i.e., <1 %), the condition frequently leads to death [1]. Surgical treatment is indicated following the failure of conservative management. Nevertheless, such patients are often characterized by poor pulmonary function and general condition. Thus, there is a need to develop effective and less invasive procedure.

Several methods are used to close the fistula from the bronchial lumen and the thoracic cavity. In endoscopic bronchial occlusion for non-operable patients with BPF, use of ethyl-2-cyanoacrylate has been advocated for smaller fistulas, while a combination of an endobronchial Watanabe spigot (EWS) and ethyl-2-cyanoacrylate has been recommend for fistulas with a larger diameter [2]. However, it is often observed that strong coughing or exertion can cause a filled EWS to migrate or be exsanguinated within a short period of time.

Silicon stents are devices originally used to treat airway stenosis. In the present case, we successfully used a one-side-closed silicone stent for the closure of a refractory tracheobronchial fistula.

2. Case report

A male patient (age; 85-years) with lung cancer underwent a right basal segmentectomy.

One week following the operation, he developed high fever and purulent sputum, which led to the diagnosis of empyema. Air leakage was detected after the insertion of a drainage tube into the thoracic cavity. Eleven days following surgery, the respiratory condition of the patient worsened. Subsequently, open-window thoracotomy was carried out. Moreover, a novel technique involving the use of an EWS, expanded polytetrafluoroethylene mesh (Gore-Tex DualMesh Biomaterial; W. L. Gore & Associates, Flagstaff, AZ, USA cjs0038), polyglycolic acid sheet, and N-butyl-2-cyanoacrylate (NBCA; Histoacryl®; Braun, Melsungen, Germany) was employed to achieve closure of the BPF [3] (Fig. 1A).

Fig. 1.

(A) Chest radiography after first treatment showing beaded EWS in Anterior basal branch (B8) (thin arrow), and in Medial basal branch (B7) (thick arrow).

(B) Chest radiography 9 months after first treatment. The beaded EWS were slightly (thin arrow) and completely (thick arrow) deviated.

(C) Coronal thoracic computed tomography imaging before second treatment. CT showed a maximum diameter of 8.7mm and a length of 15.0mm at the fistula site.

However, 9 months later, the fistula area was found to be wide open (Fig. 1B). The patient had been treated for air leaks with gauze compression, however, this approach was not sufficiently effective, making sputum expectoration and vocalization difficult. Computed tomography showed a maximum diameter and length measuring 8.7mm and 15.0mm, respectively, at the fistula site (Fig. 1C). A 20-mm long silicone stent with an outer diameter measuring 9mm was adjusted to fit the target bronchus as measured by computed tomography beforehand. Thereafter, only one of the severed ends was cut, and a total of seven stiches were used for ligation and closure using 3-0 nylon thread (Fig. 2A). The- L-type EWS was filled for the purpose of retaining the stent lumen (Fig. 2B). The customized silicone stent was grasped with forceps, guided into the responsible bronchus, and implanted through direct view from the thoracic side. The EWS was inserted and implanted from the pleural side so that the cut and closed side was the bronchial lumen side. The EWS in silicone had a semicircular projection on its body part, which provided sufficient fixation, so no adhesive was used (Fig. 3A). Post-implantation radiography also showed that the right basal segment bronchus was closed with an implanted silicone stent (Fig. 3B). Air leakage from the fistula was significantly reduced, and breathing and sputum ejection were easily achieved with light gauze compression.

Fig. 2.

(A) The procedure for stent customization. Only one of the severed ends was cut, and a total of 7 stitches were used for through ligation and closure using 3-0 nylon thread.

(B) The-L typed EWS was filled for the purpose of retaining the stent lumen.

Fig. 3.

(A) The view from the fenestration side. Stent filled with EWS(L) and EWS(M) was inserted into B8. EWS(L) was inserted into B7.

(B) Postoperative chest radiography showing stent(①), EWS(M)(②) and EWS(L)(③).

The open window has not been closed because the omentum had already been removed after surgery for colorectal cancer, and biomaterials (e.g., fillable dorsal muscle) are difficult to obtain. However, the patient requires daily gauze changes, but is in good respiratory condition, has good infection control and the patient currently being followed up as an outpatients.

3. Discussion

BPFs occur between a bronchus and pleural space, causing serious complications, which a mortality rate of 18–67 % [4]. In case of difficulty to purify the pleural cavity and remaining lungs, open window thoracotomy might be necessary.

There is no standard treatment for the closure of BPFs after fenestration; nonetheless, direct closure and filling with muscle omentum are often performed [5]. With regard to the selection of occluding material, previous studies recommended using glues or sealants for small and acute BPFs, and solid devices for large and chronic BPFs [2]. It has been shown that using an EWS effectively treats air leaks and avoids surgery in 77.6 % of patients for whom surgery is deemed difficult. Recent evidence has indicated that, a combination of NBCA with an EWS [2], extracellular matrix fistula plug [6], and endobronchial valve may be useful [7]. Recently, endoscopic bronchial occlusion has been utilized in this setting. Nevertheless, this approach has been linked to repeated dislodgement and reinsertion of the EWS, while the use of other embolic materials has been ineffective.

Three types of EWS are available, namely; 5 mm, 6 mm, and 7 mm; nonetheless, use of this device is often associated with deviation and may lead to repeated procedures due to size mismatch with the bronchus. Therefore, in this case, the method using beaded EWS, NBCA and polyglycolic acid sheets was implemented for the first time [3]. This method may be applicable to the treatment of small fistulas and showed good progress for 9 months without migration of EWS in a short period of time. However, the BPF site opened up and a new closure method was requested.

Closure of post-pneumonectomy tracheobronchial fistulas with implantation of a customized transbronchial silicon Y-stent has been previously reported [8]. Thus far, only one case of direct closure of a tracheobronchial fistula by closing one side of a straight silicon stent has been reported [9]. Due to the tubular structure of the stent, it is not suitable for fistula closure at the end of a bronchial segment, of note, it is essential to close one side of the stent. Furthermore, this is the first case which the EWS was placed into the stent to maintain its lumen.

Closure of fistulas measuring >8 mm in diameter with a conventional EWS is difficult. The minimum diameter of the straight silicon stent is 9 mm; this stent may be indicated for the closure of fistulas with a large diameter. For the prevention of deviation in stenting, it is important to select a filling with a slightly larger outer diameter than that of the fistula. In the case of retrograde insertion, it is necessary to pay close attention to the positional relationship with the pulmonary artery and damage to pulmonary tissue.

4. Conclusion

The procedure involving the use of a one-side-closed silicon stent is not typically used for the closure of a retrograde tracheobronchial fistula. Nonetheless, it may be an effective treatment option in a limited number of cases, such as when the window is open and the fistula is over a certain size.

CRediT authorship contribution statement

Masaya Tamura: Writing – original draft. Naoki Furukawa: Project administration. Marino Yamamoto: Project administration, Data curation. Ryohei Miyazaki: Project administration. Hironobu Okada: Project administration.

Ethics statement

The authors declare that appropriate written informed consent was obtained from the patients for publication of this case report and any accompanying images.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Abbreviations

EWS

endobronchial watanabe spigot (EWS)

PGA

polyglycolic acid

NBCA

N-butyl-2-cyanoacrylate

BPF

bronchopleural fistula

ePTFE:

expanded polytetrafluoroethylene

CT

computed tomography