International Journal of Pediatric Otorhinolaryngology 115 (2018) 58–60
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Case Report
Employing bioabsorbable grafts in two-stage laryngotracheal reconstruction of pediatric patient with severe subglottic stenosis and history of airway surgery
T
Elliot Lea,∗, Lenhanh Tranb a b
Duke University School of Medicine, USA Pacific Coast Medical Inc, USA
A R T I C LE I N FO
A B S T R A C T
Keywords: Airway Bioabsorbable graft Laryngotracheal reconstruction Subglottic stenosis
A 16-month old female was referred to our practice for laryngotracheal reconstruction (LTR) for acquired subglottic stenosis (SGS) diagnosed at 4 weeks of age due to prolonged intubation. She has a history of open thoracic repair of congenital tracheoesophageal fistula that was complicated by a pneumothorax and phrenic nerve paralysis. We performed a variation of an anterior and posterior cricoid split LTR with tracheal stenting in order to avoid risks of pulmonary morbidity from costochondral cartilage harvesting. We report the first LTR to use KLS Martin Resorb-XG bioabsorbable implant (poly-L-lactic acid & poly glycolic acid) as a substitute graft for autologous cartilage in a patient with severe SGS and a history of airway surgeries. The patient had an uncomplicated recovery and had a patent trachea on laryngobronchoscopy at 4, 13, and 22 months after surgery.
1. Introduction Ninety percent of pediatric cases of acquired subglottic stenosis (SGS) are a result of prolonged endotracheal intubation. The incidence of stenosis following intubation ranges from < 1% to 8.3% [1]. With a multitude of surgical approaches, appropriate choice of laryngotracheal reconstruction (LTR) technique varies depending on stenosis severity (Cotton-Myer grade) and anatomic location of stenosis (anterior, posterior or both). Autologous costochondral cartilage is the graft most commonly used to distract cricoid cartilage in LTR. It provides a sturdy support and decreases the time of stenting in patients with posterior pathologies [2]. However, costochondral cartilage harvesting requires additional surgical time and a separate chest wall incision with inherent surgical morbidities including postoperative pain that may lead to hypoventilation, pulmonary atelectasis, and infection. Other potential surgical complications are neurovascular injury, chest wall injury and pneumothorax [3]. We describe a case of LTR with bioabsorbable grafting instead of autologous cartilage. 2. Case report A 16-month-old patient was referred to our practice for airway obstruction refractory to multiple failed tracheal balloon dilations. The
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patient was born full-term and diagnosed with type C congenital tracheoesophageal fistula (TEF) 2 cm above the carina and proximal esophageal atresia located above the thoracic inlet. Thoracoscopic repair on the third day of life was converted to open repair due to a long gap between the proximal esophageal pouch and distal TEF. Esophagoplasty was completed with extensive dissection and anastomosis between the proximal esophageal pouch and the distal esophagus using interrupted PDS sutures. TEF was ligated and closed with PDS sutures. Pneumothorax and right phrenic nerve paralysis complicated the immediate postoperative period. The patient was intubated for over 20 days, failed three attempts at extubation, and was reintubated with smaller endotracheal tubes (ETT) after each attempt. The patient was observed with a distressingly weak and hoarse cry before the third emergent reintubation with a cuffless 3.0 ETT positioned above the stenosis. Emergent tracheotomy and laryngobronchoscopy performed at 4 weeks of age revealed acquired SGS with significant subglottic edema, granulation tissue, and a tracheal lumen measuring less than 2.7 mm. The child underwent five balloon dilations by another surgeon at 7, 8, 9, 12, and 13 months of age with 5 then 7 mm InspiraAir balloons. The surgeon aborted a sixth balloon dilation when the stenosis did not show significant improvement (from 2.0 mm to 2.9 mm in diameter). Laryngobronchoscopy at 16 months of age (Fig. 1) demonstrated Cotton-Myer Grade III SGS– significant circumferential fibrosis and
Corresponding author. 929 Morreene Rd, Apt A24, Durham, NC, 27705, USA. E-mail address:
[email protected] (E. Le).
https://doi.org/10.1016/j.ijporl.2018.09.013 Received 2 July 2018; Received in revised form 16 September 2018; Accepted 16 September 2018 Available online 19 September 2018 0165-5876/ © 2018 Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 115 (2018) 58–60
E. Le, L. Tran
Postoperative endoscopic reevaluation revealed a small non-obstructive tracheal granuloma that was excised without complication at 4 months, a healed tracheal airway without restenosis at 13 months, and a patent proximal trachea at 22 months (Fig. 3). Tracheotomy decannulation was delayed but successful at 22 months due to social and geographic challenges. The child remains decannulated and is thriving 37 months after LTR. 3. Discussion LTR with cartilage graft distraction remains an important surgical approach for long segment SGS. Costochondral cartilage harvesting, however, increases operative time and increases the risk of postoperative pain, surgical site infection, scarring, pneumothorax, and atelectasis with the possibility of pneumonia [3,4]. We used KLS Martin Resorb-XG bioabsorbable implants as a substitute graft to avoid cartilage harvesting. This decreased surgical time and decreased the risk of postoperative respiratory morbidities especially in the setting of a previous right open thoracic repair of TEF and right phrenic nerve paralysis. KLS Martin Resorb-X implants are currently used for craniosynostosis and orthopedic procedures [5]. The Resorb-XG implant is a polymer of 85% poly-L-lactic acid and 15% poly glycolic acid (Fig. 4). It maintains up to 80% strength at 2 months, allowing for the surgical site to heal and re-epithelialize. The Resorb-XG has the highest initial strength among the Resorb-X line, and it has a resorption time of 12–14 months. The product is thus able to support the cricoid split for an appropriate period before full hydrolysis converts the polymer into carbon dioxide and water, allowing the child's airway to grow unobstructed [6]. Because it degrades, bioabsorbable graft have a lower risk of plate migration relative to non-degradable alternatives [7]. The use of bioabsorbable implants instead of costochondral cartilage grafts is not new to LTR. Lactosorb, a poly-L-lactic acid-poly-glycolic acid implant, was used in the single-stage reconstruction of ten Cotton-Myer Grade II and III patients with no significant posterior involvement. All patients were extubated at discharge and follow-up demonstrated well-healed anterior tracheal walls. This case series demonstrated the effectiveness of Lactosorb in the reconstruction of mild to moderate subglottic stenosis [4]. Various animal models have also assessed the viability of Lactosorb in LTR [8-10]. All studies demonstrated that Lactosorb was an appropriate alternative to autologous cartilage grafts. However, these studies lacked a robust long-term follow-up as all animals were sacrificed for graft evaluation within 12 months of surgical intervention. Despite the animal models' success, bioabsorbable implant grafting in LTR has not been widely adopted in humans.
Fig. 1. Pre-operative laryngobronchoscopy demonstrating severe circumferential subglottic stenosis – Cotton-Myer Grade III.
mucosal edema obstructing approximately 90% of the airway at the subglottic level. There was 15 mm of segmental tapering tracheal stenosis proximal to the tracheal stoma with the smallest diameter measuring 2.5 mm. Surgery was coordinated when the patient and her family were able to fly back to our hospital. We performed a two-stage anterior and posterior cricoid split LTR at 23 months of age. A midline lower neck incision was made and the tracheocutaneous fistula was excised. The anterior vertical tracheal split was extended to just superior to the tracheal stenotic site. The posterior cricoid split was performed with a beaver blade to adequately enlarge the stenosis. The anterior tracheal defect was measured 5 mm wide after placement of a 7 mm Cotton-Lorenz stent to distract both the anterior and posterior cricoid. The anterior cricoid split was augmented by a 10 mm wide, 15 mm long, 0.6 mm thick KLS Martin Resorb-XG bioabsorbable implant (Fig. 2A) and secured with multiple interrupted stitches of 4-0 vicryl sutures (Fig. 2B). The tracheotomy tube was replaced. A Penrose drain was placed in the subplatysmal plane to prevent subcutaneous emphysema and removed on postoperative day 4. The patient had an uncomplicated recovery and was weaned off mechanical ventilation in one day. The Cotton-Lorenz stent was removed 3 weeks after LTR.
Fig. 2. A KLS Martin Resorb XG implant cut and molded. 2B – Anterior cricoid split distraction with Resorb-XG implant and stenosis supported by Cotton-Lorenz Stent. 59
International Journal of Pediatric Otorhinolaryngology 115 (2018) 58–60
E. Le, L. Tran
Fig. 3. Series of post-operative laryngobronchoscopic images. 3A–4 months post-op demonstrating tracheal granuloma. 3B–13 months post-op demonstrating patent airway. 3C–22 months post-op demonstrating patent airway.
for certain patients especially with existing pulmonary morbidities or lack of adequate source of cartilage graft. The literature surrounding LTR with bioabsorbable grafts in humans is lacking and deserves future investigation as an alternative to autologous costochondral cartilage grafting. Consent The patient's family gave us their consent to write about the patient's course of care and to use images of the patient's operative site. They understood that the published report would not include the child's or the family's name and would not include any patient-identifiable health information. They understood that the article may be published in a journal that is read worldwide and that the report may be published online. Declaration of interest None. Fig. 4. KLS Martin Resorb-XG implant as it appears out of packaging – pre-cut and pre-molded.
References [1] G.H. Zalzal, R.T. Cotton, Glottic and subglottic stenosis, in: M. Lesperance, P. Flint (Eds.), Cummings Pediatric Otolaryngology, Elsevier Saunders, Philadelphia, 2015, pp. 348–360. [2] R.T. Cotton, Management of subglottic stenosis, Otolaryngol. Clin. 33 (2000) 111–130. [3] G.H. Zalzal, R.T. Cotton, A.J. McAdams, The survival of costal cartilage graft in laryngotracheal reconstruction, Otolaryngol. Head Neck Surg. 94 (1986) 204–211. [4] R.C. Sprecher, Single-stage laryngotracheal reconstruction using bioabsorbable miniplates, Laryngoscope 120 (2010) 1655–1661. [5] D.C. Nguyen, A.S. Woo, S.J. Farber, G.B. Skolnick, J. Yu, S.D. Naidoo, K.B. Patel, Comparison of resorbable plating systems: complications during degradation, J. Craniofac. Surg. 28 (2017) 88–92. [6] KLS Martin, Resorb-xg. https://www.klsmartin.com/de/produkte/implantatemund-kiefer-und-gesicht/resorbierbare-osteosynthese/?L=4 (accessed 15 June 2018). [7] M.T. Mitskavich, F.L. Rimell, A.M. Shapiro, J. Christopher Post, S.B. Kapadia, Laryngotracheal reconstruction using microplates in a porcine model with subglottic stenosis, Laryngoscope 106 (1996) 301–305. [8] A.M. Klein, V.L. Graham, Y. Gulleth, D. Lafreniere, Polyglycolic acid/poly-L-lactic acid copolymer use in laryngotracheal reconstruction: a rabbit model, Laryngoscope 115 (2005) 583–587. [9] C.M. Long, S.F. Conley, A. Kajdacsy-Balia, J.E. Kerschner, Laryngotracheal reconstruction in canines: fixation of autologous costochondral grafts using polylactic and polyglycolic acid miniplates, Arch. Otolaryngol. Head Neck Surg. 127 (2001) 570–575. [10] S.E. Pearson, F. Rimell, E.B. Stelow, K. Pernell, Tracheal reconstruction with a synthetic material in a porcine model, Ann. Otol. Rhinol. Laryngol. 110 (2001) 718–722.
Our patient had significant acquired SGS after prolonged intubation and multiple tracheal dilations. Chest wall scarring after open thoracic TEF repair compromises the harvest site, and an existing right phrenic nerve paralysis increases the risk of pulmonary complications during the recovery period. This history made the patient an ideal candidate for LTR with bioabsorbable implant grafting. In addition to patients with a high risk of pulmonary complications, patients with previous costochondral cartilage harvesting or calcified rib cartilage may be well suited for bioabsorbable implants. These products are readily available, will decrease surgical time and risk of morbidity, are easily moldable to fit the defect, are structurally robust throughout the healing period, and allow for unobstructed growth as it degrades. 4. Conclusion This is the first case to describe successful anterior and posterior cricoid split LTR using Resorb-XG bioabsorbable grafts in a patient with Cotton-Myer Grade III SGS and a history of airway and thoracic surgeries. Bioabsorbable grafting eliminates the risks associated with cartilage harvesting while offering rigid support and eventual fibrous tissue replacement. This technique should be considered a viable option
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