BRIEF REPORT
Novel Treatment of a Persistent Bronchopleural Fistula Using a Customized Spigot Aditya Jindal, MD, DM, and Ritesh Agarwal, MD, DM
Summary: An abnormal communication between the
bronchus and the pleural cavity is known as a bronchopleural fistula (BPF). The incidence of postoperative BPF ranges from 1.5% to 28%, and is responsible for considerable morbidity and mortality. Various modalities have been used for treatment of BPF including surgery and endobronchial occlusion devices. The scope of therapy is limited in developing countries because of cost constraints and lack of availability of different occlusion devices. A novel method of BPF closure using a customized spigot and cyanoacrylate glue is described. Key Words: bronchopleural fistula, stent, glue, air leak, pneumonectomy, spigot (J Bronchol Intervent Pulmonol 2014;21:173–176)
A
n abnormal communication between the bronchus and the pleural cavity is known as a bronchopleural fistula (BPF).1 It is differentiated from alveolopleural fistula, which is an abnormal communication between bronchial tree and the pleural space, arising at a level distal to the segmental bronchus.2 BPF arises as a complication of various entities including necrotizing pneumonia or empyema, lung malignancies, trauma or as a postoperative complication after lobectomy, pneumonectomy, or even lung biopsy.1 The occurrence of BPF in the postpneumonectomy setting ranges from 1.5% to 28%, and is associated with considerable morbidity and mortality due to the development of superadded pneumonia and associated acute respiratory distress syndrome.3 Various methods have been devised for the treatment of this condition. These range from conservative management in the form of Received for publication May 22, 2013; accepted February 17, 2014. From the Department of Pulmonary Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India. Disclosure: There is no conflict of interest or other disclosures. Reprints: Ritesh Agarwal, MD, DM, Department of Pulmonary Medicine, Postgraduate Institute of Medical Education and Research, Sector-12, Chandigarh 160012, India (e-mails: [email protected]; [email protected]). Copyright r 2014 by Lippincott Williams & Wilkins
J Bronchol Intervent Pulmonol
intercostal tube drainage with or without suction to surgical closure to newer endobronchial modalities like endobronchial spigots, biological glue, unidirectional valves, and others.1 However, none of these modalities have been established as the treatment of choice, and most of the literature is available in the form of brief case reports. In addition, in developing countries, treatment is further compounded by financial constraints, lack of expertise, limited availability of devices, and lack of customization of occlusion devices. Herein, we describe a novel approach for treatment of a postpneumonectomy BPF using an endobronchial spigot customized from a silicon stent. CASE REPORT A 26-year-old female presented with persistent purulent expectoration 2 years following left pneumonectomy for extensive bronchiectasis. The expectoration soon became copious in amount and increased when lying on the right side. Sputum cultures were sterile and she was started on oral levofloxacin 500 mg daily. She continued to have these complaints along with constitutional symptoms; however, she never had an episode of pneumonia. On evaluation, a BPF was demonstrated on the computed tomography scan, arising at the distal end of the left main bronchial stump (Fig. 1). Stump dehiscence was noted on flexible bronchoscopy with
FIGURE 1. Computed tomography scan showing a bronchopleural fistula between the left main bronchus and the pleural cavity (arrow).
Volume 21, Number 2, April 2014
www.bronchology.com |
173
Jindal and Agarwal
J Bronchol Intervent Pulmonol
the stump size of at least 1.5 to 2 cm (Fig. 2). She was initially managed conservatively with the hope that the BPF would heal spontaneously. There was no resolution of the fistula after 1 month of follow-up. Endobronchial glue instillation with 1 mL N-butyl cyanoacrylate (Nectacryl; Dr Reddy’s Lab, India) was attempted with flexible bronchoscope to close the BPF. However, this proved unsuccessful and endobronchial therapy with fibrin glue (Tisseel; Baxter Ltd., India) was tried, which also failed. The patient’s body mass index was 15.6 kg/ m2, and she was deemed to be a poor candidate for thoracic surgery. Hence, it was decided to proceed with rigid bronchoscopy and endobronchial occlusion of the fistulous tract. However, cost constraints and lack of local availability of suitable occlusive devices forced us to devise a novel way to tackle the problem. An endobronchial silicon stent was cut to the required length and folded into a conical shape. This was then secured by means of a nonabsorbable surgical suture (Fig. 3). Two different sizes of this makeshift occlusive device were prepared. After informed consent from the patient, rigid bronchoscopy was performed under general anesthesia. The conical makeshift stent was deployed by means of a stent applicator system (Tonn applicator; Novatech, France), which is a complete system for loading and deploying silicon stents through a rigid bronchoscope. The spigot was positioned in the left main bronchus such that the proximal end of the stent was distal to the main carina (Fig. 4). This was followed by the instillation of 1 mL of N-butyl cyanoacrylate glue over the stent, to prevent stent displacement and seal any small pores (Fig. 4).
FIGURE 2. Endobronchial image showing stump dehiscence in the left main bronchus (arrow). The breakdown of the suture line is also seen (arrow head).
174 | www.bronchology.com
Volume 21, Number 2, April 2014
FIGURE 3. Customized spigot for placement in the left main bronchus made from cutting and folding the silicon stent.
Postprocedure, the patient reported significant improvement of symptoms. The follow-up chest radiograph showed resolution of the air fluid level and gradual obliteration of the left hemithorax (Fig. 5). She is symptom free at 3 months’ follow-up. DISCUSSION
Postoperative BPF has been described picturesquely as a “Damocles sword” hanging over the surgeon.4 It is associated with high mortality rates ranging from 18% to 50%.5 Postoperative BPF is classified according to the time of onset after the operation as early (1 to 7 d), intermediate (8 to 30 d), and late (>30 d).6 Early fistulas are due to surgical technical problems, whereas the late BPFs are due to inadequate healing or secondary infection of the bronchial stump. The index case fitted in the latter category. Surgical management is preferred in early BPF due to instability of mediastinal structures, whereas endoscopic and conservative management may be opted in late cases. Several risk factors have been identified for the development of postoperative BPF. These include rightsided resection, presence of residual carcinoma at the bronchial stump site, and mediastinal lymph node dissection in cases of resection for malignancy, diabetes mellitus, cirrhosis, hypoalbuminemia, steroid use, and others.1,7 None of these risk factors were observed in our patient. BPFs can present in an acute, subacute, and chronic manner. The presence of a BPF may be life threatening in case of an acute presentation, leading on to aspiration pneumonia, acute respiratory distress syndrome, and difficulty in r
2014 Lippincott Williams & Wilkins
J Bronchol Intervent Pulmonol
Volume 21, Number 2, April 2014
Novel Treatment of a Persistent BPF
FIGURE 4. Spigot placed in the left main bronchus (right). This was followed by instillation of 1 mL of cyanoacrylate glue (left).
mechanical ventilation because of the excess air leak. The diagnosis of BPF is established on a combination of clinical and radiologic parameters followed by visualization of the communication on bronchoscopy. The identification of BPF is a problem only with distal leaks, that is, alveolopleural fistula because the fistula is not visualized by flexible bronchoscopy. Computed tomography, instillation of methylene blue, and ventilation scintigraphy are required in such instances.1,8,9 Fiberoptic bronchoscopy and sequential balloon occlusion are also useful for this purpose.10 Various treatments have been tried over the years for the management of BPF. However, the treatment needs to be individualized in every case depending on the patient’s clinical condition. A wide variety of endobronchial occlusive devices have been used for this purpose. The earliest endobronchial treatments for this condition were reported in 1977, using an occlusion device (lead shots) and sealant (tissue glue).11,12
Multiple other devices have been tried since then, with variable success.9,13 Reports of endobronchial stents (both metallic and silicon) being used for treatment are available. However, the stents in these case reports were either specially designed or were used to bypass the fistula.14–16 The options available with us included glue (N-butyl cyanoacrylate and fibrin glue), spigots, and stents. The instillation of glue was tried twice but was unsuccessful; the spigots available were too small (maximum size, 9 mm) to fit the left main bronchus. Because of the problems of secretions and granulation tissue formation associated with stents, it was decided to place a customized spigot. This index report is the first to use a tracheobronchial stent in a novel way for the treatment of BPF. In conclusion, no treatment or device has been shown to be superior to the other in the treatment of BPF. Management has to be individualized as no treatment is standardized. In
FIGURE 5. Preprocedure and postprocedure chest radiographs show filling up of the postpneumonectomy space after spigot placement. r
2014 Lippincott Williams & Wilkins
www.bronchology.com |
175
Jindal and Agarwal
J Bronchol Intervent Pulmonol
addition, in developing countries, factors such as cost and local availability need to be considered. ACKNOWLEDGMENT The authors acknowledge the help of Dr Ravindra Mehta, Director, Interventional Pulmonology and Critical Care Medicine, Apollo Hospitals, Bengaluru, India, for helping them in designing and placement of the stent.
REFERENCES 1. Sarkar P, Chandak T, Shah R, et al. Diagnosis and management of bronchopleural fistula. Indian J Chest Dis Allied Sci. 2010;52:97–104. 2. Singh N, Agarwal R. Bronchopleural fistula or alveolopleural fistula? Not just semantics. Chest. 2006; 130:1948, author reply 1948-1949. 3. Andreetti C, D’Andrilli A, Ibrahim M, et al. Effective treatment of post-pneumonectomy bronchopleural fistula by conical fully covered self-expandable stent. Interact Cardiovasc Thorac Surg. 2012;14:420–423. 4. Pop D, Nadeemy AS, Venissac N, et al. Bronchopleural fistula: the Damocles sword of all pneumonectomies. Interact Cardiovasc Thorac Surg. 2011;13:107–108. 5. Tayama K, Eriguchi N, Futamata Y, et al. Modified Dumon stent for the treatment of a bronchopleural fistula after pneumonectomy. Ann Thorac Surg. 2003;75:290–292. 6. Varoli F, Roviaro G, Grignani F, et al. Endoscopic treatment of bronchopleural fistulas. Ann Thorac Surg. 1998;65:807–809. 7. Asamura H, Naruke T, Tsuchiya R, et al. Bronchopleural fistulas associated with lung cancer operations.
176 | www.bronchology.com
8.
9. 10. 11. 12. 13.
14. 15.
16.
Volume 21, Number 2, April 2014
Univariate and multivariate analysis of risk factors, management, and outcome. J Thorac Cardiovasc Surg. 1992;104:1456–1464. Ricci ZJ, Haramati LB, Rosenbaum AT, et al. Role of computed tomography in guiding the management of peripheral bronchopleural fistula. J Thorac Imaging. 2002;17:214–218. Lois M, Noppen M. Bronchopleural fistulas: an overview of the problem with special focus on endoscopic management. Chest. 2005;128:3955–3965. McManigle JE, Fletcher GL, Tenholder MF. Bronchoscopy in the management of bronchopleural fistula. Chest. 1990;97:1235–1238. Ratliff JL, Hill JD, Tucker H, et al. Endobronchial control of bronchopleural fistulae. Chest. 1977;71: 98–99. Hartmann W, Rausch V. New therapeutic application of the fiberoptic bronchoscope. Chest. 1977;71:237. Wiaterek G, Lee H, Malhotra R, et al. Bronchoscopic blood patch for treatment of persistent alveolarpleural fistula. J Bronchol Interv Pulmonol. 2013;20: 171–174. Chae EY, Shin JH, Song HY, et al. Bronchopleural fistula treated with a silicone-covered bronchial occlusion stent. Ann Thorac Surg. 2010;89:293–296. Kutlu CA, Patlakoglu S, Tasci AE, et al. A novel technique for bronchopleural fistula closure: an hourglass-shaped stent. J Thorac Cardiovasc Surg. 2009;137: e46–e47. Singh SS, Pyragius MD, Shah PJ, et al. Management of a large bronchopleural fistula using a tracheobronchial stent. Heart Lung Circ. 2007;16:57–59.
r
2014 Lippincott Williams & Wilkins
Novel Treatment of a Persistent Bronchopleural Fistula Using a Customized Spigot Aditya Jindal, MD, DM, and Ritesh Agarwal, MD, DM
Summary: An abnormal communication between the
bronchus and the pleural cavity is known as a bronchopleural fistula (BPF). The incidence of postoperative BPF ranges from 1.5% to 28%, and is responsible for considerable morbidity and mortality. Various modalities have been used for treatment of BPF including surgery and endobronchial occlusion devices. The scope of therapy is limited in developing countries because of cost constraints and lack of availability of different occlusion devices. A novel method of BPF closure using a customized spigot and cyanoacrylate glue is described. Key Words: bronchopleural fistula, stent, glue, air leak, pneumonectomy, spigot (J Bronchol Intervent Pulmonol 2014;21:173–176)
A
n abnormal communication between the bronchus and the pleural cavity is known as a bronchopleural fistula (BPF).1 It is differentiated from alveolopleural fistula, which is an abnormal communication between bronchial tree and the pleural space, arising at a level distal to the segmental bronchus.2 BPF arises as a complication of various entities including necrotizing pneumonia or empyema, lung malignancies, trauma or as a postoperative complication after lobectomy, pneumonectomy, or even lung biopsy.1 The occurrence of BPF in the postpneumonectomy setting ranges from 1.5% to 28%, and is associated with considerable morbidity and mortality due to the development of superadded pneumonia and associated acute respiratory distress syndrome.3 Various methods have been devised for the treatment of this condition. These range from conservative management in the form of Received for publication May 22, 2013; accepted February 17, 2014. From the Department of Pulmonary Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India. Disclosure: There is no conflict of interest or other disclosures. Reprints: Ritesh Agarwal, MD, DM, Department of Pulmonary Medicine, Postgraduate Institute of Medical Education and Research, Sector-12, Chandigarh 160012, India (e-mails: [email protected]; [email protected]). Copyright r 2014 by Lippincott Williams & Wilkins
J Bronchol Intervent Pulmonol
intercostal tube drainage with or without suction to surgical closure to newer endobronchial modalities like endobronchial spigots, biological glue, unidirectional valves, and others.1 However, none of these modalities have been established as the treatment of choice, and most of the literature is available in the form of brief case reports. In addition, in developing countries, treatment is further compounded by financial constraints, lack of expertise, limited availability of devices, and lack of customization of occlusion devices. Herein, we describe a novel approach for treatment of a postpneumonectomy BPF using an endobronchial spigot customized from a silicon stent. CASE REPORT A 26-year-old female presented with persistent purulent expectoration 2 years following left pneumonectomy for extensive bronchiectasis. The expectoration soon became copious in amount and increased when lying on the right side. Sputum cultures were sterile and she was started on oral levofloxacin 500 mg daily. She continued to have these complaints along with constitutional symptoms; however, she never had an episode of pneumonia. On evaluation, a BPF was demonstrated on the computed tomography scan, arising at the distal end of the left main bronchial stump (Fig. 1). Stump dehiscence was noted on flexible bronchoscopy with
FIGURE 1. Computed tomography scan showing a bronchopleural fistula between the left main bronchus and the pleural cavity (arrow).
Volume 21, Number 2, April 2014
www.bronchology.com |
173
Jindal and Agarwal
J Bronchol Intervent Pulmonol
the stump size of at least 1.5 to 2 cm (Fig. 2). She was initially managed conservatively with the hope that the BPF would heal spontaneously. There was no resolution of the fistula after 1 month of follow-up. Endobronchial glue instillation with 1 mL N-butyl cyanoacrylate (Nectacryl; Dr Reddy’s Lab, India) was attempted with flexible bronchoscope to close the BPF. However, this proved unsuccessful and endobronchial therapy with fibrin glue (Tisseel; Baxter Ltd., India) was tried, which also failed. The patient’s body mass index was 15.6 kg/ m2, and she was deemed to be a poor candidate for thoracic surgery. Hence, it was decided to proceed with rigid bronchoscopy and endobronchial occlusion of the fistulous tract. However, cost constraints and lack of local availability of suitable occlusive devices forced us to devise a novel way to tackle the problem. An endobronchial silicon stent was cut to the required length and folded into a conical shape. This was then secured by means of a nonabsorbable surgical suture (Fig. 3). Two different sizes of this makeshift occlusive device were prepared. After informed consent from the patient, rigid bronchoscopy was performed under general anesthesia. The conical makeshift stent was deployed by means of a stent applicator system (Tonn applicator; Novatech, France), which is a complete system for loading and deploying silicon stents through a rigid bronchoscope. The spigot was positioned in the left main bronchus such that the proximal end of the stent was distal to the main carina (Fig. 4). This was followed by the instillation of 1 mL of N-butyl cyanoacrylate glue over the stent, to prevent stent displacement and seal any small pores (Fig. 4).
FIGURE 2. Endobronchial image showing stump dehiscence in the left main bronchus (arrow). The breakdown of the suture line is also seen (arrow head).
174 | www.bronchology.com
Volume 21, Number 2, April 2014
FIGURE 3. Customized spigot for placement in the left main bronchus made from cutting and folding the silicon stent.
Postprocedure, the patient reported significant improvement of symptoms. The follow-up chest radiograph showed resolution of the air fluid level and gradual obliteration of the left hemithorax (Fig. 5). She is symptom free at 3 months’ follow-up. DISCUSSION
Postoperative BPF has been described picturesquely as a “Damocles sword” hanging over the surgeon.4 It is associated with high mortality rates ranging from 18% to 50%.5 Postoperative BPF is classified according to the time of onset after the operation as early (1 to 7 d), intermediate (8 to 30 d), and late (>30 d).6 Early fistulas are due to surgical technical problems, whereas the late BPFs are due to inadequate healing or secondary infection of the bronchial stump. The index case fitted in the latter category. Surgical management is preferred in early BPF due to instability of mediastinal structures, whereas endoscopic and conservative management may be opted in late cases. Several risk factors have been identified for the development of postoperative BPF. These include rightsided resection, presence of residual carcinoma at the bronchial stump site, and mediastinal lymph node dissection in cases of resection for malignancy, diabetes mellitus, cirrhosis, hypoalbuminemia, steroid use, and others.1,7 None of these risk factors were observed in our patient. BPFs can present in an acute, subacute, and chronic manner. The presence of a BPF may be life threatening in case of an acute presentation, leading on to aspiration pneumonia, acute respiratory distress syndrome, and difficulty in r
2014 Lippincott Williams & Wilkins
J Bronchol Intervent Pulmonol
Volume 21, Number 2, April 2014
Novel Treatment of a Persistent BPF
FIGURE 4. Spigot placed in the left main bronchus (right). This was followed by instillation of 1 mL of cyanoacrylate glue (left).
mechanical ventilation because of the excess air leak. The diagnosis of BPF is established on a combination of clinical and radiologic parameters followed by visualization of the communication on bronchoscopy. The identification of BPF is a problem only with distal leaks, that is, alveolopleural fistula because the fistula is not visualized by flexible bronchoscopy. Computed tomography, instillation of methylene blue, and ventilation scintigraphy are required in such instances.1,8,9 Fiberoptic bronchoscopy and sequential balloon occlusion are also useful for this purpose.10 Various treatments have been tried over the years for the management of BPF. However, the treatment needs to be individualized in every case depending on the patient’s clinical condition. A wide variety of endobronchial occlusive devices have been used for this purpose. The earliest endobronchial treatments for this condition were reported in 1977, using an occlusion device (lead shots) and sealant (tissue glue).11,12
Multiple other devices have been tried since then, with variable success.9,13 Reports of endobronchial stents (both metallic and silicon) being used for treatment are available. However, the stents in these case reports were either specially designed or were used to bypass the fistula.14–16 The options available with us included glue (N-butyl cyanoacrylate and fibrin glue), spigots, and stents. The instillation of glue was tried twice but was unsuccessful; the spigots available were too small (maximum size, 9 mm) to fit the left main bronchus. Because of the problems of secretions and granulation tissue formation associated with stents, it was decided to place a customized spigot. This index report is the first to use a tracheobronchial stent in a novel way for the treatment of BPF. In conclusion, no treatment or device has been shown to be superior to the other in the treatment of BPF. Management has to be individualized as no treatment is standardized. In
FIGURE 5. Preprocedure and postprocedure chest radiographs show filling up of the postpneumonectomy space after spigot placement. r
2014 Lippincott Williams & Wilkins
www.bronchology.com |
175
Jindal and Agarwal
J Bronchol Intervent Pulmonol
addition, in developing countries, factors such as cost and local availability need to be considered. ACKNOWLEDGMENT The authors acknowledge the help of Dr Ravindra Mehta, Director, Interventional Pulmonology and Critical Care Medicine, Apollo Hospitals, Bengaluru, India, for helping them in designing and placement of the stent.
REFERENCES 1. Sarkar P, Chandak T, Shah R, et al. Diagnosis and management of bronchopleural fistula. Indian J Chest Dis Allied Sci. 2010;52:97–104. 2. Singh N, Agarwal R. Bronchopleural fistula or alveolopleural fistula? Not just semantics. Chest. 2006; 130:1948, author reply 1948-1949. 3. Andreetti C, D’Andrilli A, Ibrahim M, et al. Effective treatment of post-pneumonectomy bronchopleural fistula by conical fully covered self-expandable stent. Interact Cardiovasc Thorac Surg. 2012;14:420–423. 4. Pop D, Nadeemy AS, Venissac N, et al. Bronchopleural fistula: the Damocles sword of all pneumonectomies. Interact Cardiovasc Thorac Surg. 2011;13:107–108. 5. Tayama K, Eriguchi N, Futamata Y, et al. Modified Dumon stent for the treatment of a bronchopleural fistula after pneumonectomy. Ann Thorac Surg. 2003;75:290–292. 6. Varoli F, Roviaro G, Grignani F, et al. Endoscopic treatment of bronchopleural fistulas. Ann Thorac Surg. 1998;65:807–809. 7. Asamura H, Naruke T, Tsuchiya R, et al. Bronchopleural fistulas associated with lung cancer operations.
176 | www.bronchology.com
8.
9. 10. 11. 12. 13.
14. 15.
16.
Volume 21, Number 2, April 2014
Univariate and multivariate analysis of risk factors, management, and outcome. J Thorac Cardiovasc Surg. 1992;104:1456–1464. Ricci ZJ, Haramati LB, Rosenbaum AT, et al. Role of computed tomography in guiding the management of peripheral bronchopleural fistula. J Thorac Imaging. 2002;17:214–218. Lois M, Noppen M. Bronchopleural fistulas: an overview of the problem with special focus on endoscopic management. Chest. 2005;128:3955–3965. McManigle JE, Fletcher GL, Tenholder MF. Bronchoscopy in the management of bronchopleural fistula. Chest. 1990;97:1235–1238. Ratliff JL, Hill JD, Tucker H, et al. Endobronchial control of bronchopleural fistulae. Chest. 1977;71: 98–99. Hartmann W, Rausch V. New therapeutic application of the fiberoptic bronchoscope. Chest. 1977;71:237. Wiaterek G, Lee H, Malhotra R, et al. Bronchoscopic blood patch for treatment of persistent alveolarpleural fistula. J Bronchol Interv Pulmonol. 2013;20: 171–174. Chae EY, Shin JH, Song HY, et al. Bronchopleural fistula treated with a silicone-covered bronchial occlusion stent. Ann Thorac Surg. 2010;89:293–296. Kutlu CA, Patlakoglu S, Tasci AE, et al. A novel technique for bronchopleural fistula closure: an hourglass-shaped stent. J Thorac Cardiovasc Surg. 2009;137: e46–e47. Singh SS, Pyragius MD, Shah PJ, et al. Management of a large bronchopleural fistula using a tracheobronchial stent. Heart Lung Circ. 2007;16:57–59.
r
2014 Lippincott Williams & Wilkins
