Original Article

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Severe Tracheobronchial Injuries: Our Experience Eva Dominguez1 Carlos De La Torre1 Alejandra Vilanova Sánchez1 Francisco Hernandez1 Ruben Ortiz1 Ane M. Andres Moreno1 Jose Luis Encinas1 Juan Vazquez1 Manuel Lopez Santamaria1 Juan Antonio Tovar1 1 Department of Pediatric Surgery, Hospital Universitario La Paz,

Madrid, Spain

Address for correspondence Eva Dominguez, MD, Department of Pediatric Surgery, Hospital Universitario La Paz, Paseo de la Castellana, 261 Madrid 28046, Spain (e-mail: [email protected]).

Abstract

Keywords

► ► ► ► ►

tracheobronchial airway injury thoracic trauma cervical trauma children

Introduction Severe tracheobronchial injuries (TBI) in children are usually traumatic or iatrogenic. However, they can also be caused by mediastinal infections that lead to critical situations. We herein report our experience in the treatment of these lesions. Methods A retrospective study was conducted for patients treated at our center from 2008 to 2014. TBI was diagnosed by imaging studies and bronchoscopy. Treatment was initially conservative (drainage of air and secretions, mechanical ventilation with minimal pressures, and an early extubation) with a limited use of surgical procedures whenever necessary. Results A total of 10 patients (7 males and 3 females) with a median age of 7.5 years (range, 3–17 years) suffered TBI. The mechanism was traumatic in six (three accidental and three iatrogenic) and mediastinal infection in four (three mycotic and one bacterial abscesses). All traumatic cases responded to conservative measures, except one iatrogenic lesion, which was surgically repaired. There were no complications or residual damages. Two patients with mediastinal infection presented with sudden cardiorespiratory arrest, one with hemoptysis caused by an arteriotracheal fistula and the other because of carinal rupture. Both died before any therapeutic measures could be taken. The other two patients were treated, one with previous extracorporeal membrane oxygenation support, underwent arterial embolization, but ultimately died, and the other one survived, but required esophagectomy and creation of a thoracostome for secondary wound closure of the bronchocutaneous fistula. Conclusion Conservative treatment with gentle respiratory support suffices in most traumatic cases of TBI. Infectious abscesses with involvement of adjacent structures sometimes require complex surgery and are life-threatening.

Introduction Tracheobronchial injuries in children are rare, accounting for less than 0.05% of lesions after chest trauma in children.1–6 Airway ruptures in blunt trauma happen due to high intraluminal pressures with a closed glottis, deceleration forces or disruption by lung traction during compression of the thorax. Other causes of damage to the tracheobronchial tree include

received May 19, 2014 accepted June 23, 2014 published online August 21, 2014

iatrogenic injuries, most frequently during endotracheal intubation, with a reported incidence of 0.005%.7 The most likely affected area is the membranous trachea, as it lacks cartilaginous support. Subcutaneous emphysema, pneumomediastinum, and pneumothorax are the clinical manifestations of air leaks into cervical virtual spaces limited by cervicothoracic fascias.8 Conservative management is advisable in small, uncomplicated injuries, while surgery, via

© 2015 Georg Thieme Verlag KG Stuttgart · New York

DOI http://dx.doi.org/ 10.1055/s-0034-1386642. ISSN 0939-7248.

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Eur J Pediatr Surg 2015;25:71–76.

M

M

F

M

M

M

M

F

3

4

5

6

7

8

9

10

17

9

6

12

3

5

4

9

6

6

Age (y)

Infectious

Infectious

Infectious

Infectious

Iatrogenic

Iatrogenic

Iatrogenic

Traumatic

Traumatic

Traumatic

Type of injury

Bacterial

Mucor

Aspergillus

Aspergillus

Neck surgery

Intubation

Intubation

Garage door

Bicycle

Bicycle

Agent

Massive hemoptysis

Acute respiratory failure Massive hemoptysis

Acute respiratory failure

Massive hemoptysis

Arteriobronchial fistula

Arteriobronchial fistula

– Direct surgical visualization

Carinal rupture

Perforation of tracheal bronchus



Bronchoscopy

Right PT þ unstable ventilation



Arteriobronchial fistula

Plain Rx films

SE þ bilateral PT þ unstable ventilation

Membranous trachea and left bronchus



CT þ bronchoscopy

PM þ bilateral PT þ severe distress

Membranous trachea

Cricothyroid membrane rupture

Bronchoscopy

SE þ PM þ right PT mild respiratory distress

Membranous trachea

Site of lesion

Direct surgical visualization

CT

SE þ PM mild respiratory distress

SE

Evaluation

Symptoms

Vascular reconstruction Bronchial repair





ECMO support Arterial embolization

Surgical evaluation Suture reinforcement Early extubation

Early extubation

Early extubation

Endotracheal intubation Bilateral pleural drains

Oxygen support Pleural drain

Oxygen support

Treatment

Esophagotracheal fistula Bronchocutaneous fistula

Exitus

Exitus

Exitus

No sequelae

No sequelae

No sequelae

No sequelae

No sequelae

No sequelae

Outcome

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Abbreviations: CT, computed tomography; ECMO, extracorporeal membrane oxygenation; PM, pneumomediastinum; PT, pneumothorax; SE, subcutaneous emphysema; y, years.

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Sex

M

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Patient

Table 1 Patient demographic, etiological, clinical, and therapeutic data

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thoracotomy or transcervical approach, depending on location of tear, is performed in large leaks, as they are potentially life-threatening situations.9 Spontaneous airway rupture due to mediastinal infections usually occurs in immunocompromised patients with severe fungal tracheobronchitis, with a very high mortality.10

Patients and Method We conducted a retrospective review of all patients with some type of airway rupture treated at our institution from January 2008 to February 2014. Information was based on the review of clinical charts and imaging. The diagnosis of tracheobronchial injury was based on clinical manifestations and confirmed by image studies (plain films or computed tomography) and/or bronchoscopy. Treatment was initially conservative (pneumothorax drainage, mechanical ventilation with minimal pressures, and early extubation) with a limited use of surgical procedures whenever necessary.

Results A total of 10 patients with tracheobronchial injury were treated at our institution since 2008. They had a median age of 7.5 years (range 3–17 years) and seven patients were male. The mechanism of injury was traumatic in six and infectious in four. ►Table 1 illustrates major demographic, etiological, clinical, and therapeutic data. Three of the six (50%) traumatic injuries were caused by home or road accidents. The agent of lesion was bicycle in two and garage door crash in one. In both bicycle accidents the mechanism of injury was cervical trauma with handlebar, and both presented with subcutaneous emphysema and mild respiratory distress. On image studies, both patients showed pneumomediastinum, and one had right pneumothorax, which was drained. One patient underwent fibrobronchoscopy, which revealed a tear in the posterior upper trachea, the other one was evaluated only by computed tomography. Both maintained respiratory stability with oxygen support, without the need for more invasive maneuvers, and were discharged in less than 7 days. The patient who suffered a garage door crash suffered a high energy trauma, which

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resulted in tear of the membranous trachea extending from the third tracheal ring down to the carina and left stem bronchus. He associated fracture of the second, third and fourth left ribs, as well as orbital and maxillary sinus fracture. He presented with severe respiratory distress, bilateral massive pneumothorax, and pneumomediastinum (►Fig. 1). The situation was managed with bilateral pleural drainage and endotracheal intubation with minimal pressures, without surgical repair. He was extubated 6 days later, after a bronchoscopic evaluation. He was discharged 1 month after the accident and is doing well 3 years later. The other three patients suffered iatrogenic tracheobronchial injuries, two during endotracheal intubation and one during a thyroglossal cyst removal surgery. Intubation was performed as an emergency procedure in one patient who presented with seizures and agitation in the context of septic shock. He presented with sudden desaturation and ventilation difficulties, along with subcutaneous emphysema and bilateral pneumothorax, some minutes after intubation (►Fig. 2). The other patient suffered the injury during intubation for an elective procedure, in which the endotracheal tube perforated a right tracheal bronchus, evidenced during bronchoscopy. Both patients were managed conservatively, with extubation in less than 48 hours, and have shown no residual lesions. The remaining patient suffered a tear of the thyrocricoid membrane during removal of a thyroglossal cyst. The injury was observed and sutured during surgery. However, subcutaneous emphysema and bleeding through the wound after coughing during the first hour of recovery, forced surgical reevaluation and suture reinforcement. He remained intubated during 24 hours and was discharged on the 3rd postoperative day. Four patients suffered a tracheobronchial rupture in the context of tracheobronchial or mediastinal severe infection. Invasive pulmonary aspergillosis affected two patients, one after bone-marrow transplantation because of acute lymphoid leukemia and the other after a multivisceral transplantation 20 months earlier. Mediastinal Mucor infection was present in a 9-year-old boy recently diagnosed with type I diabetes mellitus. All presented sudden cardiorespiratory arrest, and two with massive hemoptysis. Two patients died during the acute episode, while the patient with acute leukemia, who was previously on extracorporeal membrane

Fig. 1 Computed tomography scan (a) and bronchoscopic images (b) showing tracheal disruption (white arrow) in patient number 4.

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Fig. 2 Severe pneumomediastinum and subcutaneous emphysema.

oxygenation support, was stabilized and treated with arterial embolization. Despite all efforts, he died 24 hours later because of rebleeding. Autopsy findings showed ulcerative necrotizing tracheobronchitis in all three cases, with creation of an arteriotracheal fistula in two and carinal rupture in the other patient. The remaining patient was a 17-year old girl with a history of pulmonary artery atresia, multiple cardiac interventions and a stent in a pulmonary collateral artery. She was admitted with a bacterial aneurismatic dilatation of the stented vessel. During corrective surgery, she suffered rupture of the aneurism and left bronchus, with massive bleeding through the endotracheal tube. Bleeding was controlled and bronchus was sutured, however, due to ongoing mediastinal infection she suffered dehiscence of the bronchial suture and esophagobronchial fistula. She required esophaguectomy and creation of a thoracostome for secondary wound closure of the bronchocutaneous fistula.

Discussion Rupture or laceration of the intrathoracic tracheobronchial tree can be the result of an external trauma or iatrogenic causes. Other less frequent causes include inhalation of noxious gases, or the aspiration of foreign bodies.11 Rupture because of chronic pulmonary or mediastinal infection can be considered a different entity happening in selected patients and with a very different management and prognosis. Tracheobronchial injuries are rarely seen in cervical or thoracic pediatric trauma, and are more frequent in children above the age of 8 years. Cervical traumatisms are infrequent in smaller children because of their shorter neck and larger head, which gives the larynx and cervical trachea a protected position.12–15 The flexibility and mobility of the soft cartilages prevents from lesions in thoracic trauma. Distortion of the European Journal of Pediatric Surgery

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U-shaped tracheal rings causes tearing of the mucosa of the longitudinal trachea in a longitudinal fashion.14 It is possible that the frequency of these injuries is underestimated, as some of the most serious cases are not diagnosed owing to multiple associated injuries and death.16,17 Trauma can be either blunt or penetrating, blunt trauma being responsible for approximately 94% or lesions in children, unlike adults in whom penetrating injuries are most frequent.18 Most common causes involve motor vehicle or pedestrian accidents, as well as bicycle accidents with direct trauma from handlebars,15 such was the case in two of our patients. Iatrogenic injuries of the tracheobronchial tree are most commonly associated with intubation. Other possible mechanisms are neck or thoracic surgery, such as in tracheotomy creation, bronchoscopy, esophagectomy, and tracheal stent placement.19,20 Mechanical factors, such as repeated attempts at intubation, overdistension or rupture of cuff, and anatomic abnormalities of the trachea are considered as the main causes of this type of complication.21,22 In our series, one patient underwent a difficult intubation because of agitation, and another one had an anatomic variation with a tracheal bronchus, as a predisposing factor. Presenting signs and symptoms of tracheobronchial injuries can be quite subtle and sometimes minimal, delaying diagnosis.15 Subcutaneous emphysema is the most specific symptom of tracheal disruption. The combination of pneumothorax and mediastinal or subcutaneous emphysema strongly suggests the presence of an intrathoracic injury. These usually appear some minutes after the trauma, but can also be delayed, with reports in literature ranging up to 4 days from the occurrence of the injury and the diagnosis.23 Tension pneumothorax is also a frequent finding, present in up to 24% of the cases.18 Pneumothorax that persists on closed chest drainage or a collapsed lung that fails to expand should alert of the possibility of a major intrathoracic injury. Respiratory distress is not constant, but is obviously the determining factor in its management, as it can range from mild respiratory discomfort to severe respiratory distress needing rapid resuscitation. Hemoptysis from rupture of tracheobronchial vessels is rare but severely life threatening. Most authors agree that bronchoscopic examination is the technique that best establishes the diagnosis of tracheobronchial rupture and should not be replaced by computed tomography or other imaging techniques.11,14 On the contrary, other authors suggest that bronchoscopy might not always be useful for initial management, especially in cases of well tolerated lesions and when the diagnosis of tracheal disruption is obvious, based on symptoms, clinical examination, and radiographic images.24 Furthermore, ventilatory condition is frequently worsened by bronchoscopy as increased airway pressure at the moment of anesthetic induction may favor air leakage.24,25 We support this second opinion, with two patients in our series who did not undergo bronchoscopy. In both the cases, diagnosis was well established after plain chest films or computed tomography, and both were respiratorily stable. Bronchoscopic evaluation was considered unnecessary as it probably would not have changed patient management.

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treatment with or without endotracheal intubation with a gentle respiratory support suffices in most cases. Ruptures due to tracheobronchial or mediastinal infections usually happen in patients with severe immunodepression and, despite all surgical and medical efforts, are in most cases fatal. Conflict of Interest None.

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Conclusions Injuries to the tracheobronchial tree are rare in children, and are most frequently caused by a blunt trauma. Early diagnosis and intervention remain the most important aspects for optimal management. In our limited experience, conservative

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cases of blunt chest trauma from motor vehicle accidents. J Pediatr Surg 1992;27(5):551–555 Cooper A, Barlow B, DiScala C, String D. Mortality and truncal injury: the pediatric perspective. J Pediatr Surg 1994;29(1):33–38 Balci AE, Kazez A, Eren S, Ayan E, Ozalp K, Eren MN. Blunt thoracic trauma in children: review of 137 cases. Eur J Cardiothorac Surg 2004;26(2):387–392 Ismail MF, al-Refaie RI. Chest trauma in children, single center experience. Arch Bronconeumol 2012;48(10):362–366 Borasio P, Ardissone F, Chiampo G. Post-intubation tracheal rupture. A report on ten cases. Eur J Cardiothorac Surg 1997;12(1):98–100 Tovar JA, Vazquez JJ. Management of chest trauma in children. Paediatr Respir Rev 2013;14(2):86–91 Carbognani P, Bobbio A, Cattelani L, Internullo E, Caporale D, Rusca M. Management of postintubation membranous tracheal rupture. Ann Thorac Surg 2004;77(2):406–409 Clarke A, Skelton J, Fraser RS. Fungal tracheobronchitis. Report of 9 cases and review of the literature. Medicine (Baltimore) 1991; 70(1):1–14 Duval EL, Geraerts SD, Brackel HJ. Management of blunt tracheal trauma in children: a case series and review of the literature. Eur J Pediatr 2007;166(6):559–563 Becmeur F, Donato L, Horta-Geraud P, et al. Rupture of the airways after blunt chest trauma in two children. Eur J Pediatr Surg 2000; 10(2):133–135 Cay A, Imamoğlu M, Sarihan H, Koşucu P, Bektaş D. Tracheobronchial rupture due to blunt trauma in children: report of two cases. Eur J Pediatr Surg 2002;12(6):419–422 Corsten G, Berkowitz RG. Membranous tracheal rupture in children following minor blunt cervical trauma. Ann Otol Rhinol Laryngol 2002;111(3 Pt 1):197–199 Slimane MA, Becmeur F, Aubert D, et al. Tracheobronchial ruptures from blunt thoracic trauma in children. J Pediatr Surg 1999; 34(12):1847–1850 Bertelsen S, Howitz P. Injuries of the trachea and bronchi. Thorax 1972;27(2):188–194 Perchinsky M, Long W, Rosoff J, Campbell TJ. Traumatic rupture of the tracheobronchial tree in a 2. year old. J Pediatr Surg 1994; 29(12):1548–1549 Nakayama DK, Rowe MI. Intrathoracic tracheobronchial injuries in childhood. Int Anesthesiol Clin 1988;26(1):42–49 Cunningham LC, Jatana KR, Grischkan JM. Conservative management of iatrogenic membranous tracheal wall injury: a discussion of 2 successful pediatric cases. JAMA Otolaryngol Head Neck Surg 2013;139(4):405–410 Miñambres E, González-Castro A, Burón J, Suberviola B, Ballesteros MA, Ortiz-Melón F. Management of postintubation tracheobronchial rupture: our experience and a review of the literature. Eur J Emerg Med 2007;14(3):177–179 European Journal of Pediatric Surgery

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Small ruptures of the trachea either from trauma or as a consequence of endotracheal intubation are most frequently managed with conservative therapy.23,26,27 However, other authors find surgical repair the most prudent action in ruptures of significant size, particularly if there is any gaping of the wound edges.14 Conservative management with endotracheal intubation beyond the site of injury, preferably with spontaneous ventilation, allows for spontaneous healing of the trachea. Positive pressure ventilation should be avoided as it carries the risk of exacerbating the injury. If the patient’s condition is otherwise stable and the injury is contained, the possibility of avoiding intubation should be considered. Treatment with antibiotics is usually administered to reduce the likelihood of mediastinal infection. Conservative treatment has yielded good results in minor lacerations, with practically no long-term complications.12,14,15 Surgical management is indicated in cases of tracheal or bronchial transection, or in cases of uncontrollable air leaks, with satisfactory results in over 90% of the cases.13,15,28 Five of the six patients with traumatic or iatrogenic injuries in our series were managed conservatively, with no need for intubation in two of them. Extubation was carried as soon as possible, to avoid residual damages and scarring. Even one patient with severe laceration of the entire posterior trachea and left bronchus was able to be managed conservatively, with a fast tracheal spontaneous healing. There have been no cases of strictures because of granulation tissue in long-term follow-up. Tracheobronchitis is a different entity that can be bacterial, viral, fungal or noninfectious, caused by mechanical ventilation, such as in neonates. Fungal tracheobronchitis occurs in severely immunocompromised patients, such as those with AIDS, hematological malignancies, neoplastic disease, chronic obstructive airway disease, diabetes mellitus, organ transplant patients, and so on. Most common etiologic factor is Aspergillus, although other microorganisms such as Candida or Mucor have been described. Overall incidence of Aspergillus tracheobronchitis occurs in less than 7% of pulmonary aspergillosis cases. In lung transplant recipients, however, frequencies of 20 to 35% in the first 6 months posttransplantation have been reported.29–31 Infection can manifest as either a superficial intraluminal process resulting in pseudomembrane formation, as fungal airway plugs or as localized ulcerative plaques starting at the mucosa with later invasion of the tracheal or bronchial wall.32 Local necrosis and cartilage invasion, with perforation into the mediastinum, and hemorrhage from bronchiopulmonary fistula are described in some reports in literature, with no survivors.33,34 Three patients in our series suffered a fungal infection that ultimately caused this type of complication, all with a fatal outcome. The patient with a bacterial mediastinal aneurism survived, but suffered a very high morbidity with important chronic sequelae.

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21 Massard G, Rougé C, Dabbagh A, et al. Tracheobronchial lacerations

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after intubation and tracheostomy. Ann Thorac Surg 1996;61(5): 1483–1487 Ross HM, Grant FJ, Wilson RS, Burt ME. Nonoperative management of tracheal laceration during endotracheal intubation. Ann Thorac Surg 1997;63(1):240–242 Jougon J, Ballester M, Choukroun E, Dubrez J, Reboul G, Velly JF. Conservative treatment for postintubation tracheobronchial rupture. Ann Thorac Surg 2000;69(1):216–220 Poli-Merol ML, Belouadah M, Parvy F, Chauvet P, Egreteau L, Daoud S. Tracheobronchial injury by blunt trauma in children: is emergency tracheobronchoscopy always necessary? Eur J Pediatr Surg 2003;13(6):398–402 Kaptanoglu M, Dogan K, Nadir A, et al. Tracheobronchial rupture: a considerable risk for young teenagers. Int J Pediatr Otorhinolaryngol 2002;62(2):123–128 Hager J, Gunkel AR, Riccabona U. Isolated longitudinal rupture of the posterior tracheal wall following blunt neck trauma. Eur J Pediatr Surg 1999;9(2):104–108 Marty-Ané CH, Picard E, Jonquet O, Mary H. Membranous tracheal rupture after endotracheal intubation. Ann Thorac Surg 1995; 60(5):1367–1371

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Pediatr Surg 1991;26(11):1316–1319 29 Kemper CA, Hostetler JS, Follansbee SE, et al. Ulcerative and

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plaque-like tracheobronchitis due to infection with Aspergillus in patients with AIDS. Clin Infect Dis 1993;17(3):344–352 Husain S, Kwak EJ, Obman A, et al. Prospective assessment of Platelia Aspergillus galactomannan antigen for the diagnosis of invasive aspergillosis in lung transplant recipients. Am J Transplant 2004;4(5):796–802 Marjani M, Tabarsi P, Najafizadeh K, et al. Pulmonary aspergillosis in solid organ transplant patients: a report from Iran. Transplant Proc 2008;40(10):3663–3667 Denning DW, Follansbee SE, Scolaro M, Norris S, Edelstein H, Stevens DA. Pulmonary aspergillosis in the acquired immunodeficiency syndrome. N Engl J Med 1991;324(10):654–662 Lévy V, Burgel PR, Rabbat A, Cornet M, Molina T, Zittoun R. Respiratory distress due to tracheal aspergillosis in a severely immunocompromised patient. Acta Haematol 1998;100(2):85–87 Boettcher H, Bewig B, Hirt SW, Möller F, Cremer J. Topical amphotericin B application in severe bronchial aspergillosis after lung transplantation: report of experiences in 3 cases. J Heart Lung Transplant 2000;19(12):1224–1227

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Severe tracheobronchial injuries: our experience.

Severe tracheobronchial injuries (TBI) in children are usually traumatic or iatrogenic. However, they can also be caused by mediastinal infections tha...
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