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Endobronchial occlusion with one-way endobronchial valves: A novel technique for persistent air leaks in children
Journal of Pediatric Surgery 50 (2015) 82–85
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Endobronchial occlusion with one-way endobronchial valves: A novel technique for persistent air leaks in children Jennifer W. Toth a, Abigail B. Podany b,⁎, Michael F. Reed c, Dorothy V. Rocourt b, Christopher R. Gilbert a, Mary C. Santos b, Robert E. Cilley b, Peter W. Dillon b a b c
Division of Pulmonary, Allergy, and Critical Care Medicine, Department of Medicine, Penn State Milton S. Hershey Medical Center, Hershey, PA Division of Pediatric Surgery, Department of Surgery, Penn State Milton S. Hershey Medical Center, Hershey, PA Division of Cardiothoracic Surgery, Department of Surgery, Penn State Milton S. Hershey Medical Center, Hershey, PA
a r t i c l e
i n f o
Article history: Received 28 September 2014 Accepted 6 October 2014 Key words: Air leak Bronchopleural fistula Bronchoscopy Endobronchial valve
a b s t r a c t Purpose: In children, persistent air leaks can result from pulmonary infection or barotrauma. Management strategies include surgery, prolonged pleural drainage, ventilator manipulation, and extracorporeal membrane oxygenation (ECMO). We report the use of endobronchial valve placement as an effective minimally invasive intervention for persistent air leaks in children. Methods: Children with refractory prolonged air leaks were evaluated by a multidisciplinary team (pediatric surgery, interventional pulmonology, pediatric intensive care, and thoracic surgery) for endobronchial valve placement. Flexible bronchoscopy was performed, and air leak location was isolated with balloon occlusion. Retrievable one-way endobronchial valves were placed. Results: Four children (16 months to 16 years) had prolonged air leaks following necrotizing pneumonia (2), lobectomy (1), and pneumatocele (1). Patients had 1–4 valves placed. Average time to air leak resolution was 12 days (range 0–39). Average duration to chest tube removal was 25 days (range 7–39). All four children had complete resolution of air leaks. All were discharged from the hospital. None required additional surgical interventions. Conclusion: Endobronchial valve placement for prolonged air leaks owing to a variety of etiologies was effective in these children for treating air leaks, and their use may result in resolution of fistulae and avoidance of the morbidity of pulmonary surgery. © 2015 Elsevier Inc. All rights reserved.
Persistent air leaks represent rare and challenging clinical problems in children. Consisting of alveolopleural or bronchopleural fistulae, they can be associated with significant morbidity and increased hospital length of stay [1]. Common etiologies include necrotizing pneumonia, ruptured pneumatocele or cystic lesions, barotrauma, direct lung trauma, and following surgery. Many different strategies have been utilized to manage persistent air leaks, including chest tube manipulations, mechanical ventilation maneuvers, reoperative surgery, and extracorporeal membrane oxygenation (ECMO) in extreme cases [2]. All of these interventions have variable success rates. In adults, bronchoscopic therapies for persistent air leaks include balloon occlusion, application of fibrin glue or other biodegradable substances, and most recently, the placement of one-way endobronchial valves [1,3–9]. One-way endobronchial valves are devices placed using bronchoscopy and have been used in the management of postoperative air leaks [6,7]. In one series of adult
⁎ Corresponding author at: Penn State Hershey Medical Center; 500 University Drive, H159; Hershey, PA, 17033. Tel.: +1 575 308 9887; fax: +1 717 531 5393. E-mail address: [email protected] (A.B. Podany). http://dx.doi.org/10.1016/j.jpedsurg.2014.10.007 0022-3468/© 2015 Elsevier Inc. All rights reserved.
patients, complete resolution of air leaks was observed to be 48%, with an additional 45% of patients experiencing partial resolution. The principle of the valves is to allow mucus and trapped air to escape through the airway while preventing inspired air from entering through the bronchus into the fistula [1]. The use of endobronchial valves has not been previously reported in pediatric patients. We identified four pediatric patients with challenging air leaks from a variety of etiologies as candidates for valve placement (Table 1).
1. Materials and methods Starting in 2012, children with refractory prolonged air leaks were evaluated by a multidisciplinary team consisting of pediatric surgery, interventional pulmonology, pediatric critical care, and thoracic surgery for endobronchial valve placement. Patients were considered candidates for valve placement after exhausting all conventional nonoperative measures and were being considered for surgical intervention. Endobronchial valve placement in children was approved by the Institutional Review Board of Penn State Milton S. Hershey Medical Center under a Humanitarian Device Exemption, and this study was approved by the Institutional Review Board.
J.W. Toth et al. / Journal of Pediatric Surgery 50 (2015) 82–85
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Table 1 Patient clinical characteristics prior to valve placement. Patient Age (years) sex
Diagnosis
Chest tube duration (pre-valve)
A B
3F 14 F
Necrotizing pneumonia, bronchopleural fistula 76 Days Influenza A and B, superimposed necrotizing pneumonia, 56 Days bronchopleural fistula
C
16 F
D
1.3 M
Influenza A, superimposed necrotizing pneumonia, ARDS, secondary spontaneous pneumothorax Aspiration pneumonia, ARDS, pneumatocele, secondary spontaneous pneumothorax
Total number of chest tubes (pre-valve) 5 7
2 Days
2
7 Days
3
Mechanical ventilation
Surgical procedures
None Yes, 2 days, perioperative for VATS Yes, 10 days
VATS decortication 1. VATS decortication 2. right thoracotomy RLL lobectomy
Yes, continuous (home ventilator)
None⁎
None⁎
Abbreviations: F = female, M = male, ARDS = acute respiratory distress syndrome, RLL = right lower lobe, RUL = right upper lobe, VATS = video-assisted thoracoscopic surgery. ⁎ not counting chest tube thoracostomy.
2. Technique The IBV Valve (Spiration, Redmond, WA) consists of an umbrellashaped polyurethane membrane fixed to a nitinol frame within a delivery catheter that is passed through the working channel greater than 2.6 mm in a therapeutic flexible bronchoscope (Fig. 1A). Techniques of valve placement have been well described [10,11]. In all children, endobronchial valve placement was performed under general anesthesia. Three patients were ventilated with a single-lumen endotracheal tube and one was ventilated via tracheostomy with the cuff intermittently deflated as the bronchoscope was passed transorally. A tapered adult hybrid scope (Olympus, Center Valley, PA, BFMP160F) was used for initial identification of the affected airways by balloon occlusion of the bronchial segment contributing to the fistula in two patients. Airway sizing was then performed with the calibrated balloon, and an appropriate size valve was chosen: either a 5 mm, 6 mm, or 7 mm valve [12]. An adult therapeutic scope was then used to place the valve. Of note, in the smallest patient in this study (7.7 kg), the valve was deployed using the hybrid scope running parallel to the valve delivery catheter. Inpatients were then clinically assessed daily for cessation of air leak and with plain chest radiography. Outpatients were assessed weekly postvalve placement. Valves were removed bronchoscopically four–six weeks after placement, in accordance with current FDA guidelines, based on resolution of the air leak and patient’s clinical status. Patients had immediate postremoval chest radiography and were again imaged at six months thereafter.
3. Results Four children with persistent air leaks were treated with endobronchial valve placement. The population consisted of three females and one male ranging in age from 16 months to 16 years and weighing from 7.7 to 48.7 kg. Valve placement in all four patients was
successful in controlling the fistulae. The average time to air leak resolution was 12 days after valve placement and ranged from 0 to 39 days (Table 2). All patients tolerated valve occlusion of airways without compromise of respiratory function. Two patients were treated on an outpatient basis, one patient was discharged 14 days after valve placement, while one remained in the hospital 55 days after valve placement owing to multiple comorbid conditions. Valve dwell time ranged from 38 to 69 days, and in three children they were removed without complications. One removal has been deferred until the patient’s rehabilitation is completed. There were no complications regarding valve placement, dwell time, or removal as a result of valve placement. Patients A and B both underwent VATS decortication with tube thoracostomy for necrotizing pneumonia. Patient A developed a bronchopleural fistula, and was discharged home with a one-way drainage pneumonostomy tube (16Fr Cook drainage catheter, Bloomington, IN) that remained in place with evidence of a persistent air leak for 53 days prior to valve placement as an outpatient (Fig. 1B). There was immediate resolution of the air leak at the time of the procedure. Patient B’s tube thoracostomy demonstrated a persistent air leak, which, after two weeks necessitated thoracotomy and right lower lobectomy with one-way pneumonostomy tube drainage for three additional weeks prior to valve placement as an outpatient, with resolution of the air leak by the time of pneumonostomy tube removal. Both patients had weekly follow up and pneumonostomy tubes were withdrawn slowly over the next 38 days. Patient C was intubated and mechanically ventilated for respiratory failure secondary to influenza A infection with superimposed methicillin-sensitive Staphylococcus aureus pneumonia. Two days after intubation she developed a large pneumothorax secondary to barotrauma necessitating urgent chest tube thoracostomy, which demonstrated a large, continuous air leak. Because of her unstable condition, she was taken to the operating room where three endobronchial valves were placed with immediate complete resolution of the air leak (Fig. 1C). Had valve placement been unsuccessful, immediate operative intervention would have been undertaken. Her chest tubes were
Fig. 1. A) Endobronchial valve. B) Patient A fistulogram. C) Patient C plain chest radiography. Arrow indicates location of three right upper lobe valves immediately after placement.
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J.W. Toth et al. / Journal of Pediatric Surgery 50 (2015) 82–85
Table 2 Summary of patient courses post-valve placement. Patient
Valve placement
A B C D
5 7 6 7
mm RUL × 1 mm RLL × 1 mm RUL × 2, 7 mm RUL × 1 mm LLL × 4
Chest tube duration (postvalve) 38 38 7 10, 14, 18
Valve duration (days)
Time to air leak resolution
38 59 69 N/A
Immediate 39 Days Immediate 7 days
Length of stay (days) 0* 0* 18 164
Abbreviations: RUL = right upper lobe, RLL = right lower lobe, LLL = left lower lobe, N/A = not applicable. ** = valves placed as outpatient.
removed 7 days later, and the valves were removed uneventfully 69 days after initial placement. Valve removal was delayed 22 days to allow the patient to recover from a viral upper respiratory infection at the time of initially scheduled removal. Patient D, a 16-month-old male with a 2q chromosome deletion, underwent elective cleft palate repair. Postoperatively, he developed respiratory distress requiring intubation and subsequent hypoxemic respiratory failure necessitating tracheostomy six weeks later. He developed a pneumatocele with air leak on the left with loss of approximately one-third of his tidal volume. Four endobronchial valves were placed with significant slowing of air leak. With weaning of pressure support over the next few days, his air leak sealed and chest tubes were removed sequentially on postvalve placement days 10, 14, and 18. 4. Discussion In children, persistent alveolopleural or bronchopleural fistulae resulting in clinically challenging prolonged pulmonary air leaks can develop from infections, developmental anomalies, trauma, or surgery. Numerous strategies have been proposed to treat these air leaks with inconsistent success. In extremely difficult conditions they may require surgical intervention or ECMO support. In adults, endobronchial valve placement represents an effective technique recently developed to treat emphysema (bronchoscopic lung volume reduction) and postoperative air leaks [13]. Our experience represents the first reported use of this technology to treat fistulae in children. In this series, the etiology of the air leaks was related to parenchymal infection or high ventilator support. The mechanically ventilated patients (C and D) developed pneumothoraces and subsequent high volume air leaks that severely compromised ventilation. The two other patients had one-way valve (“Heimlich”) pleural drainage systems placed for prolonged air leak and underlying infectious diagnoses. In these two patients, endobronchial valves were placed as outpatients, thus sparing them an admission. Because they were outpatients, exact time of air leak resolution could not be determined, however both had immediate decrease in observed air leak at the time of valve placement. These patient’s chest tubes were removed at a longer interval postvalve placement than mandated by the air leak itself owing to the diagnosis of pulmonary infection. In retrospect, prolonged time of pleural drainage, number of chest tubes placed, and length of stay might have been reduced by earlier consideration for endobronchial valve placement. In each case the decision was made to pursue endobronchial valve placement after it was determined that all conventional nonoperative attempts at resolving fistulae had failed and the next step would have been operative intervention. Endobronchial valve placement was attempted with the goal of obviating the need for surgery. If it had failed, surgery would have still been an option for all four patients. Following successful control of the air leak, all patients were able to have their pleural drains removed. One has required persistent support owing to developmental concerns. Follow-up examinations of the other three have demonstrated complete resolution of pulmonary symptoms and return to normal function. Subsequent chest radiographs have demonstrated resolution of all infiltrative processes and no evidence of lobar destruction or cyst formation.
The use of endobronchial valves in children to control air leaks represents an intervention that may allow some children to avoid surgical intervention or ECMO. However, their use presents several intriguing issues related to the appropriate application of this technology. It remains to be determined what underlying causes of air leaks will respond to valve placement. Air leaks associated with congenital structural abnormalities or large acquired cystic lesions may not be appropriate candidates in which to attempt this intervention. The technical limits of valve placement also have to be determined. The smallest one-way valve at this time is 4 mm. Anatomic studies have shown that 4 mm is the average diameter of a mainstem bronchus in a 10 kg infant, although airways may be significantly larger in children with bronchopulmonary dysplasia as in the case of the smallest patient in this study [14]. In addition, there are no data on which to base the decision of how long a valve should be in place in order to ensure complete resolution and healing of the fistula. Current removal at 4–6 weeks is based upon results from the adult literature [15]. Finally, the impact of valve treatment on the longterm healing and function of the affected lung is unknown. Based on our initial experience in these four children, the use of oneway endobronchial valves can be safely and successfully performed and may be an effective alternative treatment for recalcitrant air leaks in pediatric patients. The application of this technology can be considered in centers where a multidisciplinary approach involving a team of physicians with expertise in interventional pulmonology, pediatric pulmonology, pediatric surgery, thoracic surgery, and pediatric critical care can be developed. Appendix A. Discussion Endobronchial Occlusion with One-Way Endobronchial Valves: A Novel Technique for Persistent Air Leaks in Children Presented by Abigail Podany, Hershey, PA. CYNTHIA DOWNARD (Louisville, KY) I think that is a very interesting application for some extremely challenging patients. I am wondering what criteria you use to determine the timing of removal of the device. ABIGAIL PODANY The timing of removal is something that we could definitely improve upon in the future and standardize it further. Basically these patients were being considered for re-operative surgery in the case of the one patient who had previously had a VATS and surgical intervention in the other three because they just were not healing. They had their chest tubes for prolonged periods, multiple chest tubes, so that is something we need to further define is patient selection criteria. CYNTHIA DOWNARD But then how did you decide when to go back and remove the devices? ABIGAIL PODANY The adult data shows to go back to remove the valve at 4–6 weeks based on resolution of the fistula and patient clinical status. The two patients that had theirs placed as outpatients we obviously could not follow to see exactly when the fistula resolved or they no longer had an air leak on their chest tube, so the first two had their chest tubes left in longer and also because of the underlying diagnosis of necrotizing pneumonia. That is also something we could follow and see if they do need to follow the adult data and keep them in from 4–6 weeks.
J.W. Toth et al. / Journal of Pediatric Surgery 50 (2015) 82–85
MARCUS JARBOE (Ann Arbor, MI) Did all these have single bronchopleural fistulas or did you have any with multiple, and how did you find with single balloon occlusion where the different segmental bronchuses were to put your valves? ABIGAIL PODANY If you have something like a staple line you probably have smaller air leaks rather than if you have one iatrogenic biopsy site or something. For the procedure, you systematically occlude the airways beginning with the side you do not suspect to have an air leak and make sure there is no change in the cessation or bubbling in the chest tube apparatus and then you occlude the whole other side that should be affected and you should see complete resolution and then further you can map it out to smaller and smaller airways. MARCUS JARBOE I had a kid on ECMO for 80 days and he had bad bilateral disease. We had that problem with these valves. You know, with balloon occlusion you never find the fistula because you occlude one main fistula and then you have another one and so there was never a change in the bubbling. I ended up taking him to IR and doing segmental contrast injections, filling the ones with glue that I could and then identifying the other ones for valve placement. It ended up working out all right but. UNIDENTIFIED SPEAKER One last quick question about your IRB approval process. You implied that this was compassionate use. Was that on a per patient basis? Do you convene a group to make a decision that would be an appropriate application of technology or did you have blanket approval? ABIGAIL PODANY We have blanket approval for a specific period of time. As this is a rare condition, we just thought we would go for the year and then we’ve had an extension to see if we can continue that. UNIDENTIFIED SPEAKER And what is the conversation with families when you’re introducing this as a therapeutic option to surgery? ABIGAIL PODANY It’s introduced as an investigational use. Most patients and their families at this point when they have
85
been having these conversations are kind of at their wits end having had multiple chest tubes for a prolonged time, empyema tubes that they have to take care of at home, “most” – three people.
References [1] El-Sameed Y, Waness A, Al Shamsi I, et al. Endobronchial valves in the management of broncho-pleural and alveolo-pleural fistulae. Lung 2012;190:347–51. [2] Amin R, Noone PG, Ratjen F. Chemical pleurodesis versus surgical intervention for persistent and recurrent pneumothoraces in cystic fibrosis. Cochrane Database Syst Rev 2012;12:1–9 [CD007481]. [3] al Jishi N, Dyer D, Sharief N, et al. Selective bronchial occlusion for treatment of bullous interstitial emphysema and bronchopleural fistula. J Pediatr Surg 1994;29: 1545–7. [4] Kuint J, Lubin D, Martinowitz U, et al. Fibrin glue treatment for recurrent pneumothorax in a premature infant. Am J Perinatol 1996;13:245–7. [5] Sarkar S, Hussain N, Herson V. Fibrin glue for persistent pneumothorax in neonates. J Perinatol 2003;23:82–4. [6] Travaline JM, McKenna Jr RJ, De Giacomo T, et al. Treatment of persistent pulmonary air leaks using endobronchial valves. Chest 2009;136:355–60. [7] Wood DE, Cerfolio RJ, Gonzalez X, et al. Bronchoscopic management of prolonged air leak. Clin Chest Med 2010;31:127–33. [8] Jester I, Nijran A, Singh M, et al. Surgical management of bronchopleural fistula in pediatric empyema and necrotizing pneumonia: efficacy of the serratus anterior muscle digitation flap. J Pediatr Surg 2012;47:1358–62. [9] Ruttenstock E, Saxena AK, Hollwarth ME. Closure of bronchopleural fistula with porcine dermal collagen and fibrin glue in an infant. Ann Thorac Surg 2012;94:659–60. [10] Wood DE, McKenna Jr RJ, Yusen RD, et al. A multicenter trial of an intrabronchial valve for treatment of severe emphysema. J Thorac Cardiovasc Surg 2007;133: 65–73. [11] Reed MF, Gilbert CR, Toth JW. Endobronchial valves for challenging air leaks. Presented at the 50th Annual Meeting of The Society of Thoracic Surgeons, Orlando, FL, January 25–29, 2014; 2014. [12] Mahajan AK, Doeing DC, Hogarth DK. Isolation of persistent air leaks and placement of intrabronchial valves. J Thorac Cardiovasc Surg 2013;145:626–30. [13] Sterman DH, Mehta AC, Wood DE, et al. A multicenter pilot study of a bronchial valve for the treatment of severe emphysema. Respiration 2010;79:222–33. [14] Tiddens HA, Hofhuis W, Casotti V, et al. Airway dimensions in bronchopulmonary dysplasia: implications for airflow obstruction. Pediatr Pulmonol 2008;43(12): 1206–13. [15] Gillespie CT, Sterman DH, Cerfolio RJ, et al. Endobronchial valve treatment for prolonged air leaks of the lung: a case series. Ann Thorac Surg 2011;91(1):270–3.
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Endobronchial occlusion with one-way endobronchial valves: A novel technique for persistent air leaks in children Jennifer W. Toth a, Abigail B. Podany b,⁎, Michael F. Reed c, Dorothy V. Rocourt b, Christopher R. Gilbert a, Mary C. Santos b, Robert E. Cilley b, Peter W. Dillon b a b c
Division of Pulmonary, Allergy, and Critical Care Medicine, Department of Medicine, Penn State Milton S. Hershey Medical Center, Hershey, PA Division of Pediatric Surgery, Department of Surgery, Penn State Milton S. Hershey Medical Center, Hershey, PA Division of Cardiothoracic Surgery, Department of Surgery, Penn State Milton S. Hershey Medical Center, Hershey, PA
a r t i c l e
i n f o
Article history: Received 28 September 2014 Accepted 6 October 2014 Key words: Air leak Bronchopleural fistula Bronchoscopy Endobronchial valve
a b s t r a c t Purpose: In children, persistent air leaks can result from pulmonary infection or barotrauma. Management strategies include surgery, prolonged pleural drainage, ventilator manipulation, and extracorporeal membrane oxygenation (ECMO). We report the use of endobronchial valve placement as an effective minimally invasive intervention for persistent air leaks in children. Methods: Children with refractory prolonged air leaks were evaluated by a multidisciplinary team (pediatric surgery, interventional pulmonology, pediatric intensive care, and thoracic surgery) for endobronchial valve placement. Flexible bronchoscopy was performed, and air leak location was isolated with balloon occlusion. Retrievable one-way endobronchial valves were placed. Results: Four children (16 months to 16 years) had prolonged air leaks following necrotizing pneumonia (2), lobectomy (1), and pneumatocele (1). Patients had 1–4 valves placed. Average time to air leak resolution was 12 days (range 0–39). Average duration to chest tube removal was 25 days (range 7–39). All four children had complete resolution of air leaks. All were discharged from the hospital. None required additional surgical interventions. Conclusion: Endobronchial valve placement for prolonged air leaks owing to a variety of etiologies was effective in these children for treating air leaks, and their use may result in resolution of fistulae and avoidance of the morbidity of pulmonary surgery. © 2015 Elsevier Inc. All rights reserved.
Persistent air leaks represent rare and challenging clinical problems in children. Consisting of alveolopleural or bronchopleural fistulae, they can be associated with significant morbidity and increased hospital length of stay [1]. Common etiologies include necrotizing pneumonia, ruptured pneumatocele or cystic lesions, barotrauma, direct lung trauma, and following surgery. Many different strategies have been utilized to manage persistent air leaks, including chest tube manipulations, mechanical ventilation maneuvers, reoperative surgery, and extracorporeal membrane oxygenation (ECMO) in extreme cases [2]. All of these interventions have variable success rates. In adults, bronchoscopic therapies for persistent air leaks include balloon occlusion, application of fibrin glue or other biodegradable substances, and most recently, the placement of one-way endobronchial valves [1,3–9]. One-way endobronchial valves are devices placed using bronchoscopy and have been used in the management of postoperative air leaks [6,7]. In one series of adult
⁎ Corresponding author at: Penn State Hershey Medical Center; 500 University Drive, H159; Hershey, PA, 17033. Tel.: +1 575 308 9887; fax: +1 717 531 5393. E-mail address: [email protected] (A.B. Podany). http://dx.doi.org/10.1016/j.jpedsurg.2014.10.007 0022-3468/© 2015 Elsevier Inc. All rights reserved.
patients, complete resolution of air leaks was observed to be 48%, with an additional 45% of patients experiencing partial resolution. The principle of the valves is to allow mucus and trapped air to escape through the airway while preventing inspired air from entering through the bronchus into the fistula [1]. The use of endobronchial valves has not been previously reported in pediatric patients. We identified four pediatric patients with challenging air leaks from a variety of etiologies as candidates for valve placement (Table 1).
1. Materials and methods Starting in 2012, children with refractory prolonged air leaks were evaluated by a multidisciplinary team consisting of pediatric surgery, interventional pulmonology, pediatric critical care, and thoracic surgery for endobronchial valve placement. Patients were considered candidates for valve placement after exhausting all conventional nonoperative measures and were being considered for surgical intervention. Endobronchial valve placement in children was approved by the Institutional Review Board of Penn State Milton S. Hershey Medical Center under a Humanitarian Device Exemption, and this study was approved by the Institutional Review Board.
J.W. Toth et al. / Journal of Pediatric Surgery 50 (2015) 82–85
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Table 1 Patient clinical characteristics prior to valve placement. Patient Age (years) sex
Diagnosis
Chest tube duration (pre-valve)
A B
3F 14 F
Necrotizing pneumonia, bronchopleural fistula 76 Days Influenza A and B, superimposed necrotizing pneumonia, 56 Days bronchopleural fistula
C
16 F
D
1.3 M
Influenza A, superimposed necrotizing pneumonia, ARDS, secondary spontaneous pneumothorax Aspiration pneumonia, ARDS, pneumatocele, secondary spontaneous pneumothorax
Total number of chest tubes (pre-valve) 5 7
2 Days
2
7 Days
3
Mechanical ventilation
Surgical procedures
None Yes, 2 days, perioperative for VATS Yes, 10 days
VATS decortication 1. VATS decortication 2. right thoracotomy RLL lobectomy
Yes, continuous (home ventilator)
None⁎
None⁎
Abbreviations: F = female, M = male, ARDS = acute respiratory distress syndrome, RLL = right lower lobe, RUL = right upper lobe, VATS = video-assisted thoracoscopic surgery. ⁎ not counting chest tube thoracostomy.
2. Technique The IBV Valve (Spiration, Redmond, WA) consists of an umbrellashaped polyurethane membrane fixed to a nitinol frame within a delivery catheter that is passed through the working channel greater than 2.6 mm in a therapeutic flexible bronchoscope (Fig. 1A). Techniques of valve placement have been well described [10,11]. In all children, endobronchial valve placement was performed under general anesthesia. Three patients were ventilated with a single-lumen endotracheal tube and one was ventilated via tracheostomy with the cuff intermittently deflated as the bronchoscope was passed transorally. A tapered adult hybrid scope (Olympus, Center Valley, PA, BFMP160F) was used for initial identification of the affected airways by balloon occlusion of the bronchial segment contributing to the fistula in two patients. Airway sizing was then performed with the calibrated balloon, and an appropriate size valve was chosen: either a 5 mm, 6 mm, or 7 mm valve [12]. An adult therapeutic scope was then used to place the valve. Of note, in the smallest patient in this study (7.7 kg), the valve was deployed using the hybrid scope running parallel to the valve delivery catheter. Inpatients were then clinically assessed daily for cessation of air leak and with plain chest radiography. Outpatients were assessed weekly postvalve placement. Valves were removed bronchoscopically four–six weeks after placement, in accordance with current FDA guidelines, based on resolution of the air leak and patient’s clinical status. Patients had immediate postremoval chest radiography and were again imaged at six months thereafter.
3. Results Four children with persistent air leaks were treated with endobronchial valve placement. The population consisted of three females and one male ranging in age from 16 months to 16 years and weighing from 7.7 to 48.7 kg. Valve placement in all four patients was
successful in controlling the fistulae. The average time to air leak resolution was 12 days after valve placement and ranged from 0 to 39 days (Table 2). All patients tolerated valve occlusion of airways without compromise of respiratory function. Two patients were treated on an outpatient basis, one patient was discharged 14 days after valve placement, while one remained in the hospital 55 days after valve placement owing to multiple comorbid conditions. Valve dwell time ranged from 38 to 69 days, and in three children they were removed without complications. One removal has been deferred until the patient’s rehabilitation is completed. There were no complications regarding valve placement, dwell time, or removal as a result of valve placement. Patients A and B both underwent VATS decortication with tube thoracostomy for necrotizing pneumonia. Patient A developed a bronchopleural fistula, and was discharged home with a one-way drainage pneumonostomy tube (16Fr Cook drainage catheter, Bloomington, IN) that remained in place with evidence of a persistent air leak for 53 days prior to valve placement as an outpatient (Fig. 1B). There was immediate resolution of the air leak at the time of the procedure. Patient B’s tube thoracostomy demonstrated a persistent air leak, which, after two weeks necessitated thoracotomy and right lower lobectomy with one-way pneumonostomy tube drainage for three additional weeks prior to valve placement as an outpatient, with resolution of the air leak by the time of pneumonostomy tube removal. Both patients had weekly follow up and pneumonostomy tubes were withdrawn slowly over the next 38 days. Patient C was intubated and mechanically ventilated for respiratory failure secondary to influenza A infection with superimposed methicillin-sensitive Staphylococcus aureus pneumonia. Two days after intubation she developed a large pneumothorax secondary to barotrauma necessitating urgent chest tube thoracostomy, which demonstrated a large, continuous air leak. Because of her unstable condition, she was taken to the operating room where three endobronchial valves were placed with immediate complete resolution of the air leak (Fig. 1C). Had valve placement been unsuccessful, immediate operative intervention would have been undertaken. Her chest tubes were
Fig. 1. A) Endobronchial valve. B) Patient A fistulogram. C) Patient C plain chest radiography. Arrow indicates location of three right upper lobe valves immediately after placement.
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J.W. Toth et al. / Journal of Pediatric Surgery 50 (2015) 82–85
Table 2 Summary of patient courses post-valve placement. Patient
Valve placement
A B C D
5 7 6 7
mm RUL × 1 mm RLL × 1 mm RUL × 2, 7 mm RUL × 1 mm LLL × 4
Chest tube duration (postvalve) 38 38 7 10, 14, 18
Valve duration (days)
Time to air leak resolution
38 59 69 N/A
Immediate 39 Days Immediate 7 days
Length of stay (days) 0* 0* 18 164
Abbreviations: RUL = right upper lobe, RLL = right lower lobe, LLL = left lower lobe, N/A = not applicable. ** = valves placed as outpatient.
removed 7 days later, and the valves were removed uneventfully 69 days after initial placement. Valve removal was delayed 22 days to allow the patient to recover from a viral upper respiratory infection at the time of initially scheduled removal. Patient D, a 16-month-old male with a 2q chromosome deletion, underwent elective cleft palate repair. Postoperatively, he developed respiratory distress requiring intubation and subsequent hypoxemic respiratory failure necessitating tracheostomy six weeks later. He developed a pneumatocele with air leak on the left with loss of approximately one-third of his tidal volume. Four endobronchial valves were placed with significant slowing of air leak. With weaning of pressure support over the next few days, his air leak sealed and chest tubes were removed sequentially on postvalve placement days 10, 14, and 18. 4. Discussion In children, persistent alveolopleural or bronchopleural fistulae resulting in clinically challenging prolonged pulmonary air leaks can develop from infections, developmental anomalies, trauma, or surgery. Numerous strategies have been proposed to treat these air leaks with inconsistent success. In extremely difficult conditions they may require surgical intervention or ECMO support. In adults, endobronchial valve placement represents an effective technique recently developed to treat emphysema (bronchoscopic lung volume reduction) and postoperative air leaks [13]. Our experience represents the first reported use of this technology to treat fistulae in children. In this series, the etiology of the air leaks was related to parenchymal infection or high ventilator support. The mechanically ventilated patients (C and D) developed pneumothoraces and subsequent high volume air leaks that severely compromised ventilation. The two other patients had one-way valve (“Heimlich”) pleural drainage systems placed for prolonged air leak and underlying infectious diagnoses. In these two patients, endobronchial valves were placed as outpatients, thus sparing them an admission. Because they were outpatients, exact time of air leak resolution could not be determined, however both had immediate decrease in observed air leak at the time of valve placement. These patient’s chest tubes were removed at a longer interval postvalve placement than mandated by the air leak itself owing to the diagnosis of pulmonary infection. In retrospect, prolonged time of pleural drainage, number of chest tubes placed, and length of stay might have been reduced by earlier consideration for endobronchial valve placement. In each case the decision was made to pursue endobronchial valve placement after it was determined that all conventional nonoperative attempts at resolving fistulae had failed and the next step would have been operative intervention. Endobronchial valve placement was attempted with the goal of obviating the need for surgery. If it had failed, surgery would have still been an option for all four patients. Following successful control of the air leak, all patients were able to have their pleural drains removed. One has required persistent support owing to developmental concerns. Follow-up examinations of the other three have demonstrated complete resolution of pulmonary symptoms and return to normal function. Subsequent chest radiographs have demonstrated resolution of all infiltrative processes and no evidence of lobar destruction or cyst formation.
The use of endobronchial valves in children to control air leaks represents an intervention that may allow some children to avoid surgical intervention or ECMO. However, their use presents several intriguing issues related to the appropriate application of this technology. It remains to be determined what underlying causes of air leaks will respond to valve placement. Air leaks associated with congenital structural abnormalities or large acquired cystic lesions may not be appropriate candidates in which to attempt this intervention. The technical limits of valve placement also have to be determined. The smallest one-way valve at this time is 4 mm. Anatomic studies have shown that 4 mm is the average diameter of a mainstem bronchus in a 10 kg infant, although airways may be significantly larger in children with bronchopulmonary dysplasia as in the case of the smallest patient in this study [14]. In addition, there are no data on which to base the decision of how long a valve should be in place in order to ensure complete resolution and healing of the fistula. Current removal at 4–6 weeks is based upon results from the adult literature [15]. Finally, the impact of valve treatment on the longterm healing and function of the affected lung is unknown. Based on our initial experience in these four children, the use of oneway endobronchial valves can be safely and successfully performed and may be an effective alternative treatment for recalcitrant air leaks in pediatric patients. The application of this technology can be considered in centers where a multidisciplinary approach involving a team of physicians with expertise in interventional pulmonology, pediatric pulmonology, pediatric surgery, thoracic surgery, and pediatric critical care can be developed. Appendix A. Discussion Endobronchial Occlusion with One-Way Endobronchial Valves: A Novel Technique for Persistent Air Leaks in Children Presented by Abigail Podany, Hershey, PA. CYNTHIA DOWNARD (Louisville, KY) I think that is a very interesting application for some extremely challenging patients. I am wondering what criteria you use to determine the timing of removal of the device. ABIGAIL PODANY The timing of removal is something that we could definitely improve upon in the future and standardize it further. Basically these patients were being considered for re-operative surgery in the case of the one patient who had previously had a VATS and surgical intervention in the other three because they just were not healing. They had their chest tubes for prolonged periods, multiple chest tubes, so that is something we need to further define is patient selection criteria. CYNTHIA DOWNARD But then how did you decide when to go back and remove the devices? ABIGAIL PODANY The adult data shows to go back to remove the valve at 4–6 weeks based on resolution of the fistula and patient clinical status. The two patients that had theirs placed as outpatients we obviously could not follow to see exactly when the fistula resolved or they no longer had an air leak on their chest tube, so the first two had their chest tubes left in longer and also because of the underlying diagnosis of necrotizing pneumonia. That is also something we could follow and see if they do need to follow the adult data and keep them in from 4–6 weeks.
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MARCUS JARBOE (Ann Arbor, MI) Did all these have single bronchopleural fistulas or did you have any with multiple, and how did you find with single balloon occlusion where the different segmental bronchuses were to put your valves? ABIGAIL PODANY If you have something like a staple line you probably have smaller air leaks rather than if you have one iatrogenic biopsy site or something. For the procedure, you systematically occlude the airways beginning with the side you do not suspect to have an air leak and make sure there is no change in the cessation or bubbling in the chest tube apparatus and then you occlude the whole other side that should be affected and you should see complete resolution and then further you can map it out to smaller and smaller airways. MARCUS JARBOE I had a kid on ECMO for 80 days and he had bad bilateral disease. We had that problem with these valves. You know, with balloon occlusion you never find the fistula because you occlude one main fistula and then you have another one and so there was never a change in the bubbling. I ended up taking him to IR and doing segmental contrast injections, filling the ones with glue that I could and then identifying the other ones for valve placement. It ended up working out all right but. UNIDENTIFIED SPEAKER One last quick question about your IRB approval process. You implied that this was compassionate use. Was that on a per patient basis? Do you convene a group to make a decision that would be an appropriate application of technology or did you have blanket approval? ABIGAIL PODANY We have blanket approval for a specific period of time. As this is a rare condition, we just thought we would go for the year and then we’ve had an extension to see if we can continue that. UNIDENTIFIED SPEAKER And what is the conversation with families when you’re introducing this as a therapeutic option to surgery? ABIGAIL PODANY It’s introduced as an investigational use. Most patients and their families at this point when they have
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been having these conversations are kind of at their wits end having had multiple chest tubes for a prolonged time, empyema tubes that they have to take care of at home, “most” – three people.
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