International Journal of Pediatric Otorhinolaryngology 73 (2009) 1817–1820
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Case report
Complete traumatic laryngotracheal disruption—A case report and review David F. Smith a, Sara Rasmussen b, Angela Peng c, Charles Bagwell d, Charles Johnson IIIe,* a
School of Medicine, Virginia Commonwealth University School of Medicine, Richmond, VA, United States Department of Surgery, Virginia Commonwealth University School of Medicine, Richmond, VA, United States c Department of Otolaryngology Head and Neck Surgery, Virginia Commonwealth University School of Medicine, Richmond, VA, United States d Department of Surgery, Division of Pediatric Surgery, Virginia Commonwealth University School of Medicine, Richmond, VA, United States e Department of Otolaryngology Head and Neck Surgery, Division of Pediatric Otolaryngology, Virginia Commonwealth University School of Medicine, Richmond, VA, United States b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 8 May 2009 Received in revised form 16 August 2009 Accepted 20 August 2009 Available online 9 October 2009
Blunt neck trauma is an infrequent cause of injury in the pediatric population; however, significant injury is possible even with minor trauma to the neck. The authors present the previously unreported case of a combined laryngotracheal and esophageal disruption as well as a severe laryngeal crush injury in a pediatric patient following a blunt, clothesline neck injury. Immediate management of laryngotracheal or esophageal separation is frequently discussed, but little information exists concerning long-term treatment and management of total laryngotracheal disruption and crush injuries in pediatric patients. A review of the literature is presented to address these concerns. ß 2009 Elsevier Ireland Ltd. All rights reserved.
Keywords: Laryngotracheal Esophageal Trauma Pediatric Disruption
1. Introduction External laryngeal trauma is a relatively rare event that can result in extensive damage to the structures of the neck or possibly death. Previous studies have reported variable incidences of laryngeal trauma as inpatient admissions, ranging from 1/5000 to 1/30,000 of Emergency Department visits [1,2], with a mortality rate of 15%, to as low as 1/137,000 ED visits [3], with a mortality of only 2.04%. A host of complications can result from blunt trauma to the neck including chronic airway obstruction and voice compromise [4,5]. Tracheal disruption can commonly occur with specific injury mechanisms, such as a clothesline injury to motorcyclists or the driver of a vehicle being struck by a steering wheel [6]. However, external blunt trauma to the neck can also result in injury to the esophagus [7]. Because initial symptoms can be highly variable and nonspecific, it is important to consider both tracheal and esophageal injury after blunt trauma to the neck, as well as laryngeal fractures. Changes in the management of laryngeal trauma, including earlier surgical intervention, have resulted in a decrease in complications seen with this type of injury [3], but it is still necessary to identify external laryngeal injuries early and provide the appropriate diagnostic testing and management.
Minor trauma to the trachea or larynx of the pediatric patient can cause significant injury, such as laryngotracheal disruption and laryngeal fracture [8,9]. For this reason, blunt neck injuries in the pediatric population, although occurring less frequently than penetrating neck injuries, are often more life-threatening [10]. Here, we report the case of a combined, complete laryngotracheal and esophageal avulsion as well as a severe laryngeal crush injury in a pediatric patient following a blunt, clothesline injury to the neck. These sequelae following external trauma to the neck have previously been discussed in an adult patient, but it is, to the best of our knowledge, unreported in the pediatric population. The first reported case of a combined laryngotracheal and esophageal injury occurred in a male patient who experienced a clothesline injury while traveling on an all-terrain-vehicle [11]; however, this was a 22year-old, adult patient, and this patient did not have a severe laryngeal crush injury from the impact with the cable. More importantly, information is available in the literature concerning acute management of laryngotracheal and esophageal disruption; however, little information exists concerning the long-term management for those pediatric patients following laryngotracheal separation and severe crush injuries. The authors present a discussion of the long-term considerations in these pediatric patients. 2. Case report
* Corresponding author at: Department of Otolaryngology Head and Neck Surgery, 1201 East Marshall Street, Suite 401, P.O. Box 980146, Richmond, VA 23298-0146, United States. Tel.: +1 804 828 2766; fax: +1 804 828 3495. E-mail address:
[email protected] (C. Johnson III). 0165-5876/$ – see front matter ß 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2009.08.022
The patient is a 12-year-old male who presented to Virginia Commonwealth University Health System (VCUHS) following an accident on his all-terrain motorbike. He was a helmeted driver
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Fig. 1. Clinical photograph of anterior neck: note the blunt injury and erythematous welt.
who experienced a blunt, clothesline injury to the anterior neck by striking a cable fence in a rural area while operating the bike at speeds in excess of 30 mph. There was a delayed transfer to VCUHS, and the patient was in cardiac arrest at presentation with cardiopulmonary resuscitation ongoing. From communication with pre-hospital personnel prior to the patient’s arrival regarding his condition, a blunt airway injury was suspected. Collaboration between anesthesia and trauma faculty resulted in a plan to attempt a nasotracheal intubation while the trauma surgeon prepared to do an emergent tracheostomy, as EMS had been unable to establish a definitive airway in the field. Nasotracheal intubation was successful on the first attempt, and the patient’s vital signs were restored. Physical exam revealed a large erythematous welt across the patient’s anterior neck at the level of the cricoid cartilage (Fig. 1). There was extensive subcutaneous emphysema from the mandible to the perineum. Bilateral chest tubes were placed for presumed pneumothoraces. As the patient was somewhat hemodynamically stable on pressors, he received head, c-spine, chest, and abdomen/pelvis computed tomography (CT) scans (Fig. 2A and B). These scans showed extensive intra- and retro-peritoneal air, as well as extensive subcutaneous air, with no signs of hollow viscous injury. Suspicion was high for a laryngotracheal crush injury, and otolaryngology
was consulted. Because the patient experienced a partial subluxation of C1 and C2, the neurosurgeons considered this injury too severe and the patient too unstable for immediate surgical intervention. The patient was taken to the ICU overnight for continued resuscitation and stabilization. Approximately 11 hours after admission, he was taken to the OR by otolaryngology for an urgent, planned neck exploration and tracheostomy. Direct laryngoscopy, with neck stabilization in the neutral position, confirmed that the epiglottis was intact, and the vocal cords were visualized flaccid without motion. Neck exploration was performed via a wide cervical collar incision with appropriate cervical stabilization using sandbags. Upon separation of the strap musculature, the endotracheal (ET) tube was visible in the operating field (Fig. 3). No intact thyroid cartilage could be identified except for multiple small, mucosally covered fragments. Diagnoses of cricotracheal separation and total laryngeal crush injury were made. At this point in time, further exploration of the neck revealed that the ET tube actually passed proximally through the hypopharynx, through an esophageal separation, and entered the trachea distally. Due to the patient’s relative instability, the trachea was brought out to the surface and secured, and the esophageal injury was closed primarily. A rigid laryngeal stent was placed in the area of the laryngeal crush injury, positioned with proline sutures, and secured with buttons in the anterior neck. An end tracheostoma was constructed. A gastrostomy tube was placed, and the patient’s incision was closed over multiple drains. The patient was returned to the ICU following the surgery. Following his initial surgery, management of our patient’s neck injury centered on options for definitive management. These options were presented to the patient’s parents: laryngotracheal reconstruction with a stent and t-tube with a chance of some vocal function, but probable permanent tracheostomy, or total laryngectomy with permanent end tracheostoma, as the parents had originally found on the internet. After careful consideration of all of the options, the parents elected to attempt laryngotracheal reconstruction. On post-injury day 12, the patient was taken to the OR for end-to-end primary anastomosis of the crushed larynx and trachea, laryngeal stent adjustment, and tracheostomy placement. The anastomosis was completed in a single layer with non-absorbable proline suture. A leak of the previous esophageal repair was noted, and this was repaired primarily. A flexible, rubberized stent was placed in the proximal, crushed larynx across anastomoses to prevent contracture.
Fig. 2. (A) Axial CT scan of the neck showing faint remnants of the thyroid cartilage and extensive subcutaneous air. (B) Sagittal CT of the neck showing proper placement of the ET and the presence of extensive subcutaneous air.
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Fig. 3. Clinical photograph of the surgical field during neck exploration: the ET tube is visible, and no intact thyroid cartilage can be identified except for multiple small fragments.
Twelve weeks following the patient’s initial injury, he presented for t-tube placement and removal of the laryngeal stent. On post-operative day 4 following this procedure, he was studied with a barium swallow and found to have no signs of aspiration or esophageal leak. Direct fiber optic laryngoscopy confirmed that the epiglottis was intact and that the vocal cords remained flaccid, without motion. Approximately 4 months after placement of the t-tube, it was removed without complication; however, due to the laryngeal crush injury, the patient had no adequate laryngeal airway. Dilation of the larynx has since been performed several times. Functionally, the patient remains able to tolerate an oral diet without aspiration. The patient currently has a permanent tracheostomy and extensive laryngeal scarring, but there is some functional sound production. 3. Discussion The patient presented in this case report had a combined laryngotracheal and esophageal separation and a severe laryngeal crush injury. Much information exists in the literature concerning medical and surgical management of patients with laryngotracheal disruptions. When one considers crush injuries in pediatric patients, little information is available concerning the long-term surgical management, especially in the setting of laryngotracheal and esophageal separation. The goals of management should focus on the preservation of an adequate laryngeal airway, swallowing, and communication, if possible. Through internet searches, the patient’s parents originally desired a total laryngectomy, choosing this procedure over a laryngotracheoplasty with a tracheostomy. Surgical options were discussed with the family comparing these two techniques, and the advantages of tissue preservation were emphasized. Laryngotracheoplasty with a tracheostomy was finally chosen for tissue preservation and hopefully to provide some functional use of the larynx even if surgery did not provide enough of a laryngeal airway to decannulate. However, this case clearly displays the reasons that treatment for patients with blunt injuries to the neck must be individualized and approached carefully, whether adult or a child. The goal of acute surgical management should be to conserve all viable structures. This is especially true for pediatric patients as growth may allow further reconstruction in the future, if not at the time of injury. By performing a total laryngectomy as a means of
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acutely treating the injuries, no options would remain long-term for any remaining tissue. A total laryngectomy may also be associated with a host of complications including an impaired sense of taste and smell, tracheobronchial infections, accumulation of debris around the stoma, and a total loss of laryngeal speech [12]. By preserving the larynx, one option for surgical management for this patient is laryngotracheal reconstruction; however, with a severe laryngeal crush injury and resulting scarring, this may be problematic. Laryngotracheal reconstruction is the procedure considered the standard of care for laryngotracheal stenosis/ subglottic stenosis in pediatric patients [13], but it is unstudied in severe laryngeal crush injuries. Subglottic stenosis is most commonly acquired through a history of intubation [14]; however, stenosis can develop from other types of external airway injuries, such as blunt trauma to the neck [13]. Laryngotracheal reconstruction is, by tradition, done as a multistage procedure utilizing a tracheostomy in situ [13]. The purpose of this procedure is to establish a stable airway while preserving or restoring normal laryngeal function [13]. Single-stage laryngotracheal reconstruction was developed in an attempt to avoid complications from traditional laryngotracheal reconstructions, such as accumulation of granulation tissue and infection [15–17]. The advantage of single-stage reconstruction is a reduction in laryngotracheal stenting and tracheostomy tube dependence [18]. More importantly, it has also been shown that laryngotracheal reconstructions performed in pediatric patients did not adversely effect laryngeal growth when used to correct congenital or acquired subglottic stenosis [19]. Although laryngotracheal reconstruction exists as a possible management option for our patient, this procedure is more commonly used for pediatric patients experiencing laryngotracheal stenosis [13] and is used when the thyroid cartilage is intact. However, the patient in this case report, in addition to combined laryngotracheal and esophageal avulsions, experienced a severe laryngeal crush injury. Severe crush injuries are considered nonlimited laryngeal injuries, as compared to subglottic stenosis. The extent of reconstruction necessary for correction of subglottic stenosis is certainly different than that required for repair of a total laryngeal crush injury. One future option for this type of severe injury, once full growth is obtained, is a total laryngeal transplant. Partial laryngeal transplants were used to treat laryngeal cancer; however, a recurrence of the tumors limited the use of transplantation for this purpose [20]. A total laryngeal transplantation was performed in 1998 on a patient who sustained a crush injury to the larynx 20 years prior [21]. Multiple laryngotracheal reconstructions were performed in this patient, all of them unsuccessful [12]. Laryngeal transplantation offered the best chance of preserving some quality of life in a patient whose larynx was irreparably damaged [12]. By approaching the acute management conservatively for our patient, the required anatomical space needed for a laryngeal transplant was preserved. Currently, our patient has an adequate, but tracheal, airway, communicates with a whisper, and tolerates oral diet without aspiration. Unless the patient undergoes a laryngeal transplant, he will continue to have a permanent tracheostoma. One option to consider at a later date to improve the voice further is placing a tracheoesophageal puncture (Blom-Singer) prosthesis. In conclusion, the authors present the first reported case of a combined laryngotracheal and esophageal separation, as well as a severe laryngeal crush injury, in a pediatric patient. The initial approach in pediatric patients with crush injuries to the neck should be to preserve anatomical structures. A conservative approach is merited because this allows the greatest degree of freedom for future procedures and rehabilitation. In this case, more
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options were preserved by utilizing the appropriate surgical management. References [1] J.P. Bent IIIrd, J.R. Silver, E.S. Porubsky, Acute laryngeal trauma: a review of 77 patients, Otolaryngol. Head Neck Surg. 109 (3 Pt. 1) (1993) 441–449. [2] S.D. Schaefer, The acute management of external laryngeal trauma: a 27-year experience, Arch. Otolaryngol. Head Neck Surg. 118 (6) (1992) 598–604. [3] B.S. Jewett, W.W. Shockley, R. Rutledge, External laryngeal trauma analysis of 392 patients, Arch. Otolaryngol. Head Neck Surg. 125 (8) (1999) 877–880. [4] G.S. Gussack, G.J. Jurkovich, A. Luterman, Laryngotracheal trauma: a protocol approach to a rare injury, Laryngoscope 96 (6) (1986) 660–665. [5] G. Minard, K.A. Kudsk, M.A. Croce, J.A. Butts, R.S. Cicala, T.C. Fabian, Laryngotracheal trauma, Am. Surg. 58 (3) (1992) 181–187. [6] M.H. Wu, Y.F. Tsai, M.Y. Lin, I.L. Hsu, Y. Fong, Complete laryngotracheal disruption caused by blunt injury, Ann. Thorac. Surg. 77 (4) (2004) 1211–1215. [7] R.J. Jones, P.C. Samson, Esophageal injury, Ann. Thorac. Surg. 19 (2) (1975) 216– 230. [8] R.P. Lusk, The evaluation of minor cervical blunt trauma in the pediatric patient, Clin. Pediatr. (Phila.) 25 (9) (1986) 445–447. [9] I.H. Kielmovitch, W.H. Friedman, Lacerations of the cervical trachea in children, Int. J. Pediatr. Otorhinolaryngol. 15 (1) (1988) 73–78. [10] H.R. Ford, M.J. Gardner, J.M. Lynch, Laryngotracheal disruption from blunt pediatric neck injuries: impact of early recognition and intervention on outcome, J. Pediatr. Surg. 30 (2) (1995) 331–334, discussion 334–335.
[11] R.A. Bernat, J.M. Zimmerman, W.M. Keane, E.A. Pribitkin, Combined laryngotracheal separation and esophageal injury following blunt neck trauma, Facial Plast. Surg. 21 (3) (2005) 187–190. [12] M. Strome, J. Stein, R. Esclamado, D. Hicks, R.R. Lorenz, W. Braun, et al., Laryngeal transplantation and 40-month follow-up, N. Engl. J. Med. 344 (22) (2001) 1676– 1679. [13] S.J. Boardman, Albert DM, Single-stage and multistage pediatric laryngotracheal reconstruction, Otolaryngol. Clin. North Am. 41 (5) (2008) 947–958, ix. [14] R.T. Cotton, J.N. Evans, Laryngotracheal reconstruction in children: five-year follow-up, Ann. Otol. Rhinol. Laryngol. 90 (5 Pt. 1) (1981) 516–520. [15] R.P. Lusk, S. Gray, H.R. Muntz, Single-stage laryngotracheal reconstruction, Arch. Otolaryngol. Head Neck Surg. 117 (2) (1991) 171–173. [16] A.B. Seid, S.M. Pransky, D.B. Kearns, One-stage laryngotracheoplasty, Arch. Otolaryngol. Head Neck Surg. 117 (4) (1991) 408–410. [17] R.T. Cotton, C.M. Myer III, D.M. O’Connor, M.E. Smith, Pediatric laryngotracheal reconstruction with cartilage grafts and endotracheal tube stenting: the singlestage approach, Laryngoscope 105 (8 Pt. 1) (1995) 818–821. [18] L.M. Gustafson, B.E. Hartley, J.H. Liu, D.T. Link, J. Chadwell, C. Koebbe, et al., Singlestage laryngotracheal reconstruction in children: a review of 200 cases, Otolaryngol. Head Neck Surg. 123 (4) (2000) 430–434. [19] N. Agrawal, M. Black, G. Morrison, Ten-year review of laryngotracheal reconstruction for pediatric airway stenosis, Int. J. Pediatr. Otorhinolaryngol. 71 (5) (2007) 699–703. [20] P. Kluyskens, S. Ringoir, Follow-up of a human larynx transplantation, Laryngoscope 80 (8) (1970) 1244–1250. [21] M. Birchall, Human laryngeal allograft: shift of emphasis in transplantation, Lancet 351 (9102) (1998) 539–540.