The Laryngoscope C 2015 The American Laryngological, V

Rhinological and Otological Society, Inc.

Case Report

Pediatric Airway Reconstruction With a Prefabricated Auricular Cartilage and Radial Forearm Free Flap Faisal I. Ahmad, MD; Karla O’Dell, MD; Jessica J. Peck, MD; Mark K. Wax, MD; Henry A. Milczuk, MD Prefabricated composite free flaps for complex airway reconstruction have been described for an adult series at our institution. We extended this approach to a pediatric patient with lifelong subglottic stenosis who had failed previous open airway reconstructions. A staged procedure was utilized in which a composite graft was created using conchal cartilages and a radial forearm free flap. This reconstruction improved the patency of her airway and decreased her dependency on intermittent airway dilations. Airway reconstruction with prefabricated conchal cartilage composite free flaps may be used as a salvage procedure for complex pediatric airway reconstruction when other methods have failed. Key Words: Pediatric, microvascular, sublottic stenosis, graft free tissue transfer. Laryngoscope, 125:1979–1982, 2015

INTRODUCTION Congenital subglottic stenosis is described as narrowing of the cricoid cartilage, whereas acquired stenosis can result from prolonged intubation or after inflammatory processes in the subglottis. Regardless of the etiology, the degree of stenosis, anatomic location and physical characteristics of the stenosis are the most important considerations for making treatment decisions.1,2 Treatment ranges from endoscopic procedures for low-grade stenosis to more invasive open procedures for higher-grade stenosis.3 Alternatively, some prefer endoscopic procedures for mucosal disease, while utilizing open procedures when there is a cartilage framework abnormality. Endoscopic procedures are commonly used as an adjunct after open procedures and sometimes concurrently in a complimentary fashion.4 Multiple open procedures have been described,5 and the selection depends on a variety of factors including the degree of stenosis, the length of the stenosis, the availability of

From the Department of Otolaryngology–Head & Neck Surgery (F.I.A., M.K.W., H.A.M.), Oregon Health & Science University, Portland, Oregon; Department of Otolaryngology–Head & Neck Surgery (K.O.), University of Southern California, Los Angeles, Los Angeles, California; the Department of Otolaryngology–Head & Neck Surgery (J.J.P.), Dwight D. Eisenhower Army Medical Center, Fort Gordon, Georgia, U.S.A. The authors have no funding, financial relationships, or conflicts of interest to disclose. Presented at the American Society of Pediatric Otolaryngology Spring Meeting, Las Vegas, Nevada, U.S.A., May 16, 2014. Received October 28, 2014 Editor’s Note: This Manuscript was accepted for publication December 11, 2014. Send correspondence to Henry A. Milczuk, MD, Department of Otolaryngology–Head and Neck Surgery, Oregon Health and Science University, 3181 SW Sam Jackson Park Road, PV01, Portland, Oregon 97239. E-mail: [email protected] DOI: 10.1002/lary.25128

Laryngoscope 125: August 2015

native tissue/airway for reconstruction, and surgeon preference and expertise. Nevertheless, open surgical management of subglottic stenosis has an estimated failure rate of 10% to 20%.6 Although reasons for failure after primary airway surgery have been described, little literature exists about further management for recurrent airway obstruction after failed open surgery. Reconstruction for patients who have undergone multiple open procedures becomes exceedingly difficult due to distorted anatomy, poor local tissue health, and the lack of local tissue. Airway reconstruction with microvascular free flaps using both autologous7 and prosthetic8 composite grafts has been previously described in the adult population. Detwiller et al.9 demonstrated that a delayed prefabricated graft using auricular cartilage and a radial forearm free flap was a viable reconstructive option in an adult series with complex laryngotracheal stenosis. This method allowed for transfer of a vascularized cartilage graft in patients who had unsuccessful previous airway reconstructions. We describe the first use of this approach in a pediatric patient with lifelong subglottic stenosis, who had failed several previous open airway reconstructions.

CASE REPORT Our patient is an 11-year-old female with a history of airway problems that developed shortly after birth. She was diagnosed with a subglottic hemangioma at 3 weeks of age. She was started on glucocorticoid therapy but was unable to tolerate continued therapy due to growth retardation and glucocorticoid-induced Cushing’s syndrome. At 3 months of age, she underwent submucosal resection of her hemangioma through an anterior cricoid split approach. She developed further airway stenosis and required multiple CO2 laser excisions and Ahmad et al.: Free Flap Pediatric Airway Reconstruction

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Fig. 1. Preoperative endoscopy. Views of supraglottic (A) and subglottic (B) airway (Cotton-Myer grade III). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]

eventually tracheostomy placement. At 2 years of age she underwent single-stage laryngotracheal reconstruction with anterior costal cartilage graft, but improvements in her airway were short-lived. A cricotracheal resection with anterior tracheoplasty with costal cartilage graft was then performed when she was 8 years of age. She then developed a near circumferential stenosis at the anastomotic site. Endoscopic images of her airway (Cotton-Myer grade III) prior to this reconstruction are shown in Figure 1. Subsequently, she had many episodes of respiratory distress requiring multiple endoscopic balloon dilations with laser scar excision and steroid injections at 3- to 4-week intervals. We had concerns that her airway would not be able to support increasing demands as she went through her adolescent growth. The frequency of procedures was negatively affecting her ability to participate in school activities. A staged composite free-tissue transfer was chosen for her reconstruction, as it brought healthy cartilage with its own blood supply to the region and provided the best chance of long-term stability. In the first stage, bilateral conchal cartilages were harvested using a subperichondrial dissection and implanted into the forearm (Fig. 2). The cartilages were arranged such that the concave sides were used to recreate with lumen of the airway, and the fasciocutaneous components could serve as coverage. Due to the expected length of native airway that would need to be resected, we elected to use both conchal cartilages. Six weeks later, the second stage was performed. Surgical approach to the airway demonstrated extensive thick scar and fibrosis, which was in excess of what we would have anticipated even in the case of a multiplyoperated field. The laryngotracheal complex was dissected from the superior margin of the thyroid cartilage to the level of the manubrium. The area of most significant stenosis and malacia, which coincided with a 3- 3 2-cm area of subglottic larynx and proximal trachea on the left anterior surface, was resected. The composite free flap was harvested concurrently. The free-tissue transfer was designed over a palpable Laryngoscope 125: August 2015

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radial artery and dissection was performed in a subfascial plane such that the conchal cartilages were raised in continuity with the free flap. The composite graft was sutured directly to the native trachea and inset such that the concave aspects of the conchal cartilages served to recreate the lumen of the airway. The free flap served as coverage over the sites of anastomoses. Microvascular anastomosis was made between the flap vessels and facial artery and vein. She remained nasotracheally intubated, and on postoperative day 6 an airway evaluation was performed. An endoscopic view of her airway is shown in Figure 3A. She was extubated the following day. She did well postoperatively, with an improvement in her breathing and better ability to engage in activities. Subsequently, she developed some airway narrowing and mild dyspnea. This was amenable to airway dilations and steroid injections and mitomycin-C application, the frequency of which had decreased significantly to every 8

Fig. 2. Creation of the composite free flap. Implanting the conchal cartilages at the donor sites. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]

Ahmad et al.: Free Flap Pediatric Airway Reconstruction

Fig. 3. Postoperative endoscopy. Views of the area of composite free flap reconstruction at 1 week (A) and 11 months (B) after surgery. The reconstructed airway (Cotton-Myer grade I) demonstrates increased luminal cross-sectional area, some mild side-toside restriction in diameter, good remucosalization, and minimal granulation tissue. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]

weeks for the first 6 months after surgery. Intraoperatively, granulation tissue was noticed at the anastomotic site, but the degree of granulation tissue formation has decreased with every subsequent procedure. Since then, her airway has appeared to have stabilized and she has only required a single balloon dilation with CO2 laser excision at 11 months postoperatively. An endoscopic image of her airway 11 months after surgery is given in Figure 3B (Cotton-Myer grade I).

DISCUSSION Our patient first developed airway problems during infancy due to a subglottic hemangioma. She did not tolerate steroid therapy, which required us to proceed with open reconstructive procedures. It is possible that her long-term use of steroids might have negatively affected wound healing after her first procedure, as high doses and prolonged use has been shown to adversely affect wound healing and is thought to prevent proper graft vascularization and mucosalization.6 Endoscopic procedures were required between the open surgeries to maintain her airway, and despite the various procedures used, her recalcitrant airway problems persisted. Her history of extensive neck surgery and failure with free-cartilage grafts limited her reconstructive Laryngoscope 125: August 2015

options. In traditional procedures, with free-cartilage transfers to augment the airway, the overlying strap musculature is critical in providing early blood supply to the avascular graft.6 Though she had some remaining musculature, the area immediately adjacent to the airway was involved with extensive fibrosis and scar tissue. More specifically, we were concerned that this local environment may have had relatively poor blood supply and may not be able to support another free-cartilage graft. A cervical slide tracheoplasty was also considered as a salvage procedure, but we did not believe there was sufficient airway length that could be sacrificed for this procedure. Consequently, we utilized a free-tissue transfer for her reconstruction. This reconstruction was performed without tracheostomy due to patient and family preference, and because of the success we had in our adult series. We selected auricular cartilage for the reconstruction because of the ease of harvest and its natural curvature, which approximates the tracheal rings, without requiring further carving or shaping. Although perichondrium was not preserved on the luminal side, the graft demonstrated good mucosalization. Additionally, by staging the procedure and first implanting the cartilage at the donor site, the thin auricular cartilage was able to develop a local blood supply prior to implantation. Prefabricated composite cartilage and radial forearm free flaps have been demonstrated as a viable and versatile option for complex adult laryngotracheal reconstruction,9 and its use in the pediatric population appears to be a logical extension. A two-team surgical approach mitigates some of the additional time added to the procedure from flap harvest and microvascular anastomosis. Although this procedure is associated with increased operative resource utilization, postoperative utilization is similar to traditional approaches with regard to intensive care unit and hospital stay. Accordingly, this approach essentially combines the tenets of airway framework reconstruction with a cartilage graft with well-established microvascular techniques. As with traditional methods, the cartilage serves as a framework to produce a wide lumen and support the native cartilage. The radial forearm free flap serves as a wellvascularized substrate to support the auricular cartilage. In most cases of airway reconstruction, the surrounding tissues can provide enough vascularization to prevent wound complications including contraction and stenosis. However, reconstruction with a composite free flap addresses the challenges of pediatric airway reconstruction in a subset of patients where there is not sufficient cartilage length and there is poor native blood supply.

CONCLUSION This case report complements the current literature on airway reconstruction with composite free flaps and demonstrates its applicability in pediatric airway management. We propose that airway reconstruction with prefabricated composite free flaps may be used as a salvage procedure for complex pediatric airway reconstruction in patients who are not candidates for traditional Ahmad et al.: Free Flap Pediatric Airway Reconstruction

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reconstruction methods or when other methods have failed.

BIBLIOGRAPHY 1. Werkhaven JA, Beste D. Diagnosis and management of pediatric laryngeal stenosis. Otolarygnol Clin North Am 1995;28:797–808. 2. Myer CM, O’Connor DM, Cotton RT. Proposed grading system for subglottic stenosis based on endotracheal tube sizes. Ann Otol Rhinol Laryngol 1994;103:319–323. 3. Brigger MT, Boseley ME. Management of tracheal stenosis. Curr Opin Otolaryngol Head Neck Surg 2012;20:491–496.

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4. Rutter MJ, Cohen AP, de Alarcon A. Endoscopic airway management in children. Curr Opin Otolaryngol Head Neck Surg 2008;16:525–529. 5. Koempel JA, Cotton RT. History of pediatric laryngotracheal reconstruction. Otolaryngol Clin North Am 2008;41:825–835. 6. de Alarcon A, Rutter MJ. Revision pediatric laryngotracheal reconstruction. Otolaryngol Clin North Am 2008;41:959–980. 7. Delaere PR, Hardillo J, Hermans R, Van Den Hof B. Prefabrication of composite tissue for improved tracheal reconstruction. Ann Otol Rhinol Laryngol 2001;110:849–860. 8. Yu P1, Clayman GL, Walsh GL. Long-term outcomes of microsurgical reconstruction for large tracheal defects. Cancer 2011;117:802–808. 9. Detwiller KY, Schindler JS, Schneider DS, Lindau R, Wax MK. Complex adult laryngotracheal reconstruction with a prefabricated flap: a case series. Head Neck 2013;35:E376–E380.

Ahmad et al.: Free Flap Pediatric Airway Reconstruction

Pediatric airway reconstruction with a prefabricated auricular cartilage and radial forearm free flap.

Prefabricated composite free flaps for complex airway reconstruction have been described for an adult series at our institution. We extended this appr...
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