The closure of complex tracheocutaneous fistulae is a surgical challenge. We describe a staged approach for management of a patient with a large tracheocutaneous fistula in the setting of prior surgery and local radiation therapy

Moore, Michael G.

Royer, Allison K.

Royer, Mark C.

Ting, Jonathan Y.

Weisberger, Edward C.

Journal of Medical Case Reports

PubMed

ScholarWorks  IU Indianapolis

The use of a prefabricated radial forearm free flap for closure of a large tracheocutaneous fistula: a case report and review of the literature

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CASE REPORT Open AccessThe use of a prefabricated radial forearm freeflap for closure of a large tracheocutaneousfistula: a case report and review of the literatureAllison K. Royer1, Mark C. Royer1, Jonathan Y. Ting2, Edward C. Weisberger2 and Michael G. Moore2*AbstractIntroduction: The closure of complex tracheocutaneous fistulae is a surgical challenge. We describe a stagedapproach for management of a patient with a large tracheocutaneous fistula in the setting of prior surgery andlocal radiation therapy.Case presentation: A 66-year-old Caucasian man who had undergone prior surgery and radiation therapy to thelower neck presented to our hospital for treatment of a large tracheocutaneous fistula that had developed with anadjacent area of tracheal stenosis. A prefabricated composite graft made up of an inner mucosal lining (buccalmucosa), a central cartilage structure (conchal cartilage), and external skin lining was constructed on the patient’sdistal volar forearm and subsequently harvested in a staged fashion. This graft was transferred as a free flap andsuccessfully used to close the patient’s defect following revascularization. Sixty months after surgery, the patienthad no airway compromise or new dysphonia.Conclusions: The use of a prefabricated mucosally lined composite graft can allow for successful closure of largetracheocutaneous fistulae, even in the setting of prior radiation therapy.Keywords: Radial forearm free flap, Tracheocutaneous fistulaeIntroductionTracheocutaneous fistulae (TCF) typically form assequelae of tracheostomy tube placement. Squamousepithelium migrates into the trachea, creating an epithe-lialized tract that fails to close following tube removal.Factors such as treatment with radiation therapy andprolonged tracheostomy tube placement increase thelikelihood of fistula formation. Problems associated withTCF include increased possibility of respiratory tract in-fections, difficulty with phonation, coughing, cosmeticproblems, and limitations on swimming and bathing,among others.Although management of smaller TCF is oftensafely and successfully accomplished by performinglimited local procedures, closure of large tracts (e.g.,greater than 4 mm) can lead to a greater risk ofperioperative complications such as subcutaneous em-physema, pneumothorax, and respiratory compromise[1]. Moreover, when the defect involves a large com-ponent of the circumference and structural support ofthe anterior trachea, the adequacy of the repaired air-way must be taken into consideration [2–5].We present a case report of a successful three-stagedrepair of a large TCF in a patient who had undergoneprior tracheal surgery and radiation therapy.Case presentationA 66-year-old Caucasian man presented to our hospitalwith a history of thyroid lymphoma treated with chemo-radiation therapy (60 Gy). Subsequent surveillancerevealed a new lesion within the thyroid gland, andworkup demonstrated a localized papillary thyroid car-cinoma treated with total thyroidectomy. Postopera-tively, he developed left true vocal fold paralysis due toclose adherence of the primary tumor to the nerve, and4 months later he developed an acute episode of airwaydistress, presumably caused by laryngeal edema, that* Correspondence: mooremg@iupui.edu2Department of Otolaryngology-Head and Neck Surgery, Indiana UniversitySchool of Medicine, 550 North University Boulevard, Room 3170,Indianapolis, IN 46202, USAFull list of author information is available at the end of the articleJOURNAL OF MEDICALCASE REPORTS© 2015 Royer et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.Royer et al. Journal of Medical Case Reports  (2015) 9:251 DOI 10.1186/s13256-015-0728-zrequired an emergent tracheostomy. The laryngealedema gradually resolved, and his tracheostomy tubewas successfully downsized and he was decannulated.He was then noted to have both a small tracheoesopha-geal fistula as well as a large TCF. The tracheoesopha-geal fistula was successfully managed with asternocleidomastoid muscle rotational flap. Bronchos-copy revealed a TCF that was 1.4 cm × 1.6 cm in sizeand had an adjacent 1.0-cm section of proximal trachealstenosis (Fig. 1). Given his history of radiation, failure ofprevious primary and rotational graft closure, and thesize and complexity of the TCF, a decision was made torepair the defect using a three-stage approach.The initial stage involved implanting a conchal cartil-age graft with the concave portion facing superficially onthe ulnar aspect of the distal left forearm. Two monthslater, owing to the patient’s dense hair distribution overthe ulnar forearm, a second-stage procedure was per-formed to place a buccal mucosal graft over the conchalcartilage graft after removing the hair-bearing skin. Afterallowing 6 weeks for healing, the third stage was per-formed. This stage involved transferring the radial fore-arm free flap (RFFF) with cartilage and mucosal graft forclosure of the TCF and augmentation of the associatedtracheal stenosis (Fig. 2).During this procedure, the conchal cartilage coveredby mucosa was inset into the tracheal defect with inter-rupted and vertical mattress 2-0 polydioxanone sutures,followed by application of a layer of fibrin glue. Supportwas augmented with a 1.2-mm absorbable miniplate(LactoSorb; W. Lorenz Surgical, Jacksonville, FL, USA).Two of these plates were used to secure the free flapgraft to a trachea (one superiorly and one inferiorly). Anarrow strip of the distal flap was deepithelialized toallow for the remaining skin to be used in the externalskin closure. Additional RFFF soft tissue was positionedbetween the tracheal closure and skin. The radial arterywas anastomosed to the right superior thyroid artery,and the cephalic vein was anastomosed to the rightexternal jugular vein. Penrose drains were placed in theneck (Fig. 3).The patient was extubated in the operating room fol-lowing reconstruction. The patient was advised to avoidstraining, speaking, and coughing in the immediate post-operative period. He had no issues with breathing orwound healing in the immediate postoperative period orat 6, 12, 18, and 60 months postoperatively.DiscussionAlthough the occurrence of TCF is not uncommon inthe pediatric population (11–43 % of the time followingdecannulation) [6–8], the literature is fairly sparse onthe occurrence of TCF in adults. Although small TCFare often successfully managed with simple tract exci-sion and/or layered closure, large TCF often require amore definitive procedure, as it is necessary not only toclose the defect but also to reconstruct the missing com-ponent of the tracheal wall.Various methods have been described for closure oflarge TCF. In short-segment lesions, when the healingcharacteristics of the wound bed are appropriate, a tra-cheal resection and end-to-end anastomosis or slidingtracheoplasty can be performed. When patient factors(e.g., diabetes mellitus, chronic lung disease, malnutri-tion, or chronic steroid use) or treatment factors (e.g.,prior neck irradiation) limit wound healing, the risks ofsuch an operation often outweigh the potential benefits.In such instances, surgeons began to look for local orregional solutions to the problem.In 1934, Jackson and Babcock described using a lined,bipedicled flap reinforced with conchal cartilage for clos-ure [9]. Authors of several subsequent case reports andcase series have described various techniques involvingdifferent types of flaps, sometimes combined with ribcartilage for structural support [10–12]. All of thesetechniques had small sample sizes with variable follow-up. Whether the patients in these reports had been pre-viously irradiated was not always discussed.More significant tracheal replacement and reconstruc-tion are a challenge for any surgeon. An excellent over-view on the subject is provided in a review by Grillo[13], a pioneer in tracheal surgery. Belsey emphasizedFig. 1 a The patient’s tracheocutaneous fistula defect included the entire anterior tracheal wall and a portion of the bilateral sidewalls. b Abronchoscopic view shows the tracheocutaneous fistula with an endotracheal tube passed through it. Proximal tracheal stenosis is evidentRoyer et al. Journal of Medical Case Reports  (2015) 9:251 Page 2 of 4requirements for successful tracheal replacement, suchas a repair that is laterally rigid but longitudinally flex-ible as well as a surface epithelium that is ciliated re-spiratory epithelium, if possible [14]. The lateralrequirement has been shown to be desirable but not es-sential [15, 16]. Substances used should be biocompat-ible and/or inert, nontoxic, noncarcinogenic, and, if notused along with immunomodulating agents, nonimmu-nogenic [17, 18].Numerous reports in recent years have described useof fasciocutaneous flaps with costal cartilage as struc-tural support [2–4]. Although this support techniqueprovides a large amount of rigid grafting material, itsharvest includes multiple risks (such as pneumothorax,hemothorax, and injury to intercostal neurovascularbundle) as well as postoperative morbidity (chest wallpain leading to atelectasis). Moreover, its straight shapemakes reconstruction of the anterior tracheal ring morechallenging, unless the cartilage is fragmented. Thisapproach is well suited for long anterior wall defects butmay not be necessary for shorter repairs.Our technique represents a novel approach in apreviously irradiated patient who had undergone mul-tiple neck surgeries before TCF closure. As a result,we needed to combine the principles of TCF repair(removal of the epithelial tract, minimizing surroundingdissection, achieving a watertight closure, and usingpassive drains) and tracheal repair (minimize woundtension and positive pressure across the repair in thepostoperative period). In our patient, the bankedmucosally lined cartilage served as a framework forstructural support of the trachea, augmented with anabsorbable reconstructive miniplate. The cartilagecurvature allowed for reconstruction of the anteriortracheal wall in a manner more natural than wouldhave been provided by costal cartilage. Moreover, themorbidity of the chest wall defect was avoided, allow-ing for easier maintenance of lung reexpansion in thepostoperative period. The use of the RFFF as a graftingmaterial provided a source of healthy, nonirradiatedtissue and additional support between the trachealclosure and the skin. The use of passive drains andvoice rest helped minimize stress on the tracheal anas-tomosis to allow for an airtight seal to be achieved.ConclusionsLarge TCF in previously irradiated patients present asurgical challenge. This case illustrates successful closureof an extensive TCF involving the anterior trachea andpartial bilateral sidewalls. The composite graft allowedfor structural support with hairless mucosal lining withina fasciocutaneous flap. This surgical technique and post-operative management strategy can be considered inpatients who present with similar TCFs.Fig. 2 a The conchal cartilage covered with buccal mucosa and implanted in the radial forearm is shown before the start of stage 3. b The radialforearm free flap with the mucosa-lined cartilage graft and skin paddle following harvest during stage 3Fig. 3 The radial forearm free flap was inset, and the radial artery and cephalic veins were anastomosed to the right superior thyroid artery andexternal jugular vein, respectively. a The mucosa-lined cartilage was positioned over the tracheal defect and secured. b The subcutaneous tissueand skin of the radial forearm free flap served as additional tissue support in final wound closure. Drains were placed in the neckRoyer et al. Journal of Medical Case Reports  (2015) 9:251 Page 3 of 4ConsentWritten informed consent was obtained from the patientfor publication of this case report and any accompanyingimages. A copy of the written consent is available forreview by the Editor-in-Chief of this journal.AbbreviationsRFFF: Radial forearm free flap; TCF: Tracheocutaneous fistulae.Competing interestsThe authors declare that they have no competing interests.Authors’ contributionsMGM was the primary surgeon, developed the surgical technique, andassisted in manuscript drafting and revision. AKR assisted with manuscriptdrafting. JYT and MCR were involved in the manuscript revisions. ECWassisted with the surgery and with manuscript revisions. All authors readand approved the final manuscript.Author details1Columbus ENT and Allergy, Columbus Regional Health, Columbus, IN, USA.2Department of Otolaryngology-Head and Neck Surgery, Indiana UniversitySchool of Medicine, 550 North University Boulevard, Room 3170,Indianapolis, IN 46202, USA.Received: 2 June 2015 Accepted: 7 October 2015References1. Drezner DA, Cantrell H. Surgical management of tracheocutaneous fistula.Ear Nose Throat J. 1998;77(7):534–7.2. Shinohara H, Yuzuriha S, Matsuo K, Kushima H, Kondoh S. Trachealreconstruction with a prefabricated deltopectoral flap combined withcostal cartilage graft and palatal mucosal graft. Ann Plast Surg.2004;53(3):278–81.3. Fujiwara T, Nishino K, Numajiri T. Tracheal reconstruction with aprefabricated and double-folded radial forearm free flap. J PlastReconstr Aesthetic Surg. 2009;60:790–4.4. Teng MS, Malkin BD, Urken ML. Prefabricated composite free flaps fortracheal reconstruction: a new technique. Ann Otol Rhinol Laryngol.2005;114(11):822–6.5. Yu P, Clayman GL, Walsh GL. Human tracheal reconstruction with acomposite radial forearm free flap and prosthesis. Ann Thorac Surg.2006;81:714–6.6. Tasca RA, Clarke RW. Tracheocutaneous fistula following paediatrictracheostomy—a 14-year experience at Alder Hey Children’s Hospital.Int J Pediatr Otorhinolaryngol. 2010;74(6):711–2.7. Mahadevan M, Barber C, Salkeld L, Douglas G, Mills N. Pediatric tracheostomy:17 year review. Int J Pediatr Otorhinolaryngol. 2007;71:1829–35.8. Joseph HT, Jani P, Preece JM, Bailey CM, Evans JN. Paediatric tracheostomy:persistent tracheo-cutaneous fistula following decannulation. Int J PediatrOtorhinolaryngol. 1991;22(3):231–6.9. Jackson C, Babcock WW. Plastic closure of tracheocutaneous fistula.Surg Clin North Am. 1934;14:199–221.10. Bishop JB, Bostwick J, Nahai F. Persistent tracheostomy stoma. Am J Surg.1980;140(5):709–10.11. Lee UJ, Goh EK, Wang SG, Hwang SM. Closure of large tracheocutaneousfistula using turn-over hinge flap and V-Y advancement flap. J LaryngolOtol. 2002;116(8):627–9.12. Riedel F, Goessler UR, Grupp S, Bran G, Hormann K, Verse T. Managementof radiation-induced tracheocutaneous tissue defects by transplantationof an ear cartilage graft and deltopectoral flap. Auris Nasus Larynx.2006;33(1):79–84.13. Grillo HC. Tracheal replacement: a critical review. Ann Thorac Surg.2002;73:1995–2004.14. Belsey R. Resection and reconstruction of the intrathoracic trachea. Br J Surg.1950;38:200–5.15. Edgerton MT, Zovickian A. Reconstruction of the trachea and infraglotticlarynx. Plast Reconstr Surg. 1954;13:167–92.16. Grillo HC. Circumferential resection and reconstruction of the mediastinaland cervical trachea. Ann Surg. 1965;162:374–88.17. Jackson TL, O’Brien EJ, Tuttle W, Meyer J. The experimental use ofhomogenous tracheal transplants in the restoration of continuity of thetracheobronchial tree. J Thorac Surg. 1950;20:598–612.18. Scherer MA, Ascherl R, Geissdörfer K, Mang W, Blümel G, Lichti H, et al.Experimental bioprosthetic reconstruction of the trachea. ArchOtorhinolaryngol. 1986;243(4):215–23.Submit your next manuscript to BioMed Centraland take full advantage of: • Convenient online submission• Thorough peer review• No space constraints or color ﬁgure charges• Immediate publication on acceptance• Inclusion in PubMed, CAS, Scopus and Google Scholar• Research which is freely available for redistributionSubmit your manuscript at www.biomedcentral.com/submitRoyer et al. Journal of Medical Case Reports  (2015) 9:251 Page 4 of 4

Allison K. Royer

Mark C. Royer

Jonathan Y. Ting

Edward C. Weisberger

Michael G. Moore

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The use of a prefabricated radial forearm free flap for closure of a large tracheocutaneous fistula: a case report and review of the literature

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