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TRACHEOINNOMINATE ARTERY FISTULA AS A COMPLICATION OF RADIATION THERAPY DAVID REITER, DMD, MD BRUCE R. PICCONE PHltlP UnMAN, MD SHElDON

A. USKER, MD

ALL BY INVITATION

PHILADElPHIA, PENNSYLVANIA

Tracheoinnomlnate artery fistulization Is a well-known complication of tracheostomy and of tracheal resection. The first known occurrence of this problem In a patient In whom no transtracheal procedure had ever been performed is reported, and high-dose radiation therapy delivered three yean before for a mediastinal malignancy Is suggested as the cause. No evidence of tumor was found In or adjacent to the tracheonscular communication. The tracheolnnomlnate artery fistula must be considered a potential complication of radiation therapy as well as of surgery.

INTRODUCTION SINCE Korte' first described the problem in 1879, tracheoinnomlnate artery fistula has become a well-known clinical entity. Long considered a complication only of tracheostomy, it has recently been de-

Submitted for publication Sept 10. 1978. From the Department of Otorhinolaryngology and Human Communication, University of Pennsylvania School of Medicine (Dr Reiter), the Section of HematologylOncology, Graduate Hospital of Philadelphia (Drs Piccone and Lisker), and the Department of Radiation Therapy, Hospital of the University of Pennsylvania (Dr Littman). Presented at the 1978 Annual Meeting of the American Academy of Otolaryngology, las Vegas, Sept1()' 13. Reprint requests to the Hospital of the University of Pennsylvania, 5 Silverstein, 3400 Spruce St, Philadelphia, PA 19174 (Dr Reiter).

scribed in patients who have undergone resection of stenotic tracheal segrnents.O To date, however, no report has appeared in the literature describing this problem in a patient who has had neither transtracheal surgery nor local neoplasm. CASE REPORT

A 46-year-old man had a mediastinal mass on a routine chest radiograph in June 1974. There were no physical symptoms. Tomography confirmed a right paratracheal mass, and whole body gallium scanning showed only mediastinal activity. Preoperative evaluation included a hemogram, coagulation survey, SMA-20, excretory urogram, and bone marrow biopsy, all of which were normal. Bronchoscopy was also normal, and mediastinoscopy revealed a right paratracheal mass, which was biopsied. The specimen was believed to be malignant but was not definitively characterized because of the limited specimen obtained. The patient underwent a right thoracotomy on June 27, 1974. The mass was partially encapsulated and adherent to the right side of the trachea. Because the judgment of several consultants was divided between malignant thymoma and anaplastic carcinoma, it was decided to deliver 6,000 rads to the mediastinum. On Aug 8, 1974, a split course of radiation therapy was begun with a 6,000 MEV linear accelerator. Three thousand rads was delivered In ten treatments to the mediastinum and supraclavicular areas using parallel opposed fields. After a five-week rest, another 3,000 rads was delivered in ten treatments using a three-field technique. The patient experienced mild dysphagia and tolerated the treatment well.

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He remained totally asymptomatic and returned to heavy cigarette smoking and his previous lifestyle and occupation. Chest radiographs, taken three times yearly, were normal until May 1977, when mild cardiomegaly and fibrosis in the mediastinum were noted. In autumn 1977 he experienced two episodes of epistaxis, each requiring nasal mucous membrane cautery, according to available records. On Nov 28, 1977, the patient was hospitalized because of acute dyspnea, productive cough, fever, and wheezing. History revealed symptoms of an upper respiratory tract infection, which had been treated conservatively by the patient's family physician for two weeks prior to admission. Physical examination showed a pulse of 112 regular beats per minute, a respiratory rate of 34, and an oral temperature of 38.5 C. Marked induration and telangiectasia were present on the chest wall and neck within the portals of radiation. Inspiratory and expiratory stridor were present, and the expiratory phase of respiration was prolonged. A chest radiograph showed tracheal narrowing with marked stenosis at the level of the carina, as well as prominent right middle lobe interstitial radio-opacity. Arterial blood gases were satisfactory, and the patient was placed on high humidity oxygen supplementation, intermittent positive pressure ventilation, and bronchodilators. Adrenal corticosteroids were started on the second hospital day, but bronchoscopy was deferred because of the concern over further tracheal narrowing following instrumentation and the difficulty anticipated in performing tracheotomy through Indurated tissue Into a trachea narrowed to beyond the carina. Significant clinical improvement was observed on the third hospital day. A barium swallow obtained on the fifth day of hospitalization revealed a general decrease in caliber of the esophagus, with a smooth area of marked stenosis at the level of the tracheal bifurcation. fiberoptic esophagoscopy done by a consultant In gastroenterology revealed only mucosal atrophy, and no biopsy specimens were taken. Mild, self-limited epistaxis was reported on the tenth hospital day. The next morning, the patient experienced massive hemoptysis, resulting in a blood loss of about 1,200 cc within ten minutes. Hemorrhage ceased without intervention, and the patient was transferred to the Intensive care unit while preparations were made for aortic arch arteriography. Hemoptysis soon recurred, during which angiography demonstrated the presence of tracheolnnomlnate artery communication. Cardiopulmonary

arrest occurred while the patient was on the angiography table, and successful resuscitation was followed by transfusion and emergency thoracotomy. Massive mediastinal fibrosis was encountered, hemorrhage was uncontrollable, and the patient died in the operating room. Postmortem examination revealed marked fibrosis of the anterior mediastinum, erosive tracheitis, and tracheoinnominate artery fistula. No evidence of neoplasia was found in any area of the trachea, innominate artery, or mediastinum.

DISCUSSION

The surgical literature contains approximately 150 reports of tracheoinnominate artery fistula. 2-s Each of these patients had undergone at least one surgical procedure in which the anterior tracheal wall was traversed, 137 for tracheostomy and the others for tracheal resection and primary repair. Hemorrhage consistently followed the precipitating procedure, lending some degree of predictability to the development of the problem. A high index of suspicion and an understanding of predisposing factors, presentation, and management are necessary for physicians involved with susceptible patient populations. Investigations into the nature of the process and predisposing factors have centered on the tracheostomized patient, as this has been widely thought to be the only susceptible population. In a study of patients having cuffed tracheostomy tubes, it was found that hypotension was associated with a greater degree of tracheal damage at the site of cuff pressure than was found in normotensive patientsP Other predisposing factors to tracheal damage from cuffed tubes include tracheal infection,8 adrenal corticosteroid administration,' and sep5is.10 The concept of lowered mucosal resistance is stressed throughout the literature on tracheoinnominate artery fistula. This manifests itself as altered response to local injury of tracheal mucosa, as from

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TRACHEOINNOMINATE ARTERY FISTULA tracheostomy tube tip or cuff. Fistulization follows injury via an intermediate phase of inflammation and fibrosis.! Late postradiation changes include vascular sclerosis, with resulting tissue ischemia and fibrosis.11-n Decreased resistance to trauma, with resultant poor healing, follows such radiation injury. The degree of change in a given patient will depend on the dose and rate of administration of radiation, but may vary widely from patient to patient because of individual factors not well understood. The chain of events leading to tracheoinnominate artery fistula begins with local tracheal injury and is connected to massive hemorrhage through impaired tissue defenses and extension of the inflammatory process into the adjacent vasculature. Prediposing factors include hypotension, corticosteroid administration, and sepsis. Diagnosis of the condition is obvious only to those knowledgeable about its existence. Sentinel bleeding, often small amounts over short periods of time, will frequently presage dramatic hemorrhage. Hypoxia may result in dark blood mistaken for venous hemorrhage. In patients who have been tracheotomized, exploration through an extension of the horizontal tracheotomy incision may be employed without sternotomy, in order to demonstrate the presence of a flstula.! Comparison of various diagnostic modalities in small series of cases suggests that no single method is reliable in the detection of a tracheovascular communicatlon.s Bronchoscopy, aortic arch arteriogram, retrograde brachial arteriogram, and wound exploration were all employed on various reported patients, with success rates ranging from 0% to 20%. Although Coopers proposes aortic arch arteriography as the diagnostic procedure of choice, Donahoo points out the subtle nature of positive angiographic findings in cases of surgically confirmed tracheo-

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innominate artery fistula, in the discussion of Cooper's work. Management includes maintenance of a secure airway, immediate control of hemorrhage, and intravascular volume replenishment. Stabilization of the patient must be achieved in order to safely proceed with operative treatment. The surgical approach to the problem should include partial sternotomy for access and resection of the involved segment of artery.' In the discussion of this work, Edie favors the individual ligation of the right carotid and subclavian arteries with resection of their origins to be included with the innominate segment. Grillo adds the need for resection of the stenotic tracheal segment in continuity with the fistula, in cases where an indwelling cuffed tube precipitated tracheal damage and tracheovascular fistulization. Saphenous vein bypass grafts have been employed for neovascularization, but the need for this procedure remains uncertain. Neurologic deficit is reportedly uncommon following interruption of the innominate artery. COMMENT Radiation injury brings about changes in mediastinal tissues favorable to the development of tracheoinnominate artery fistula. Where the response is severe and dramatic, as in the case presented, it is reasonable to expect poor results from tracheal or esophageal injury. An erosive tracheitis, probably infectious in origin, was the precipitating factor in this patient. Other considerations include the possibility of endoscopic trauma initiating tracheal ulceration in the case of bronchoscopy, or esophageal damage in the case of esophagoscopy. The literature contains no report of tracheoinnominate artery fistula following mediastinoscopy. It is not reasonable to implicate this procedure in this patient,

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for several reasons. The procedure was carried out more than three years before fistualization became apparent and more than two weeks before the initiation of radiation therapy. While some degree of scarring and fibrosis must have occurred secondary to the passage of the mediastinoscope between the trachea and the innominate artery, the presence of a fistula and the lack of bleeding into the mediastinum proper support the idea of an intratracheal origin for the penetrating process. The implications of this hypothesis are clear. Patients exposed to tumoricidal doses of ionizing radiation must be viewed with suspicion of vascular misadventure in the affected area. With mediastinal radiation, therefore, there is a possibility of subsequent tracheoinnominate artery fistualization. Upper respiratory tract infections must be treated with the utmost respect. Early culturing will facilitate identification of bacterial pathogens and their prompt eradication with appropriate antibiotics. Endoscopic procedures must be considered with regard to the balance of potential benefit and potential harm. Endotracheal intubation should be reserved for urgent or emergent procedures, and the airway should be manipulated as gently and briefly as possible. Where sentinel bleeding has been observed and the diagnosis is confirmed by angiography or subsequent massive hemorrhage, endotracheal intubation by mouth will secure the airway and aid in control of the bleeding. The inflated cuff will tamponade most of the bleeding, aiding in stabilization for surgical intervention. While exploration of the site through a preexisting tracheotomy wound may have some merit in the nonradiated patient, it is not advisable to perform a tracheotomy in a patient in order to secure the airway or explore the area. Emergency tracheotomy is hazardous enough in normal tissues and healthy

patients with foreign bodies of the upper airway, and is far less safe and practical than orotracheal intubation under the circumstances being discussed.

SUMMARY A case of tracheoinnominate artery fistula in a patient having neither tracheotomy nor neoplasm at the site has been presented. The primary cause was believed to be severe postradiation change in the tissues of the mediastinum, with the precipitating event being an upper respiratory infection. There is a potential for this problem in a population not previously thought to be susceptible. Reasonable precautions are proposed for the expectant management of such patients, and guidelines are offered for therapy, should the need arise.

REFERENCES 1. Korte W: Ueber einige seltenere Nachkrankheiten nach der Tracheotomie wegen Oiptheritis. Arch Klin Chir 24:238, 1879. 2. Revilla AG Jr, Donahoo JS, Cameron Il.: Tracheal-innominate artery fistula after tracheal reconstruction: A case of successful repair. J Thorac Cardiovasc Surg 67:629-633, 1974. 3. Arbulu A, Thoms NW: Tracheal-innominate artery fistula after repair of tracheal stenosis: Problem and prevention. J Thorac Cardiovasc Surg 67:936-940, 1974. 4. jones jW, Reynolds M, Hewitt RL, et al: Tracheo-innominate artery erosion: Successful surgical management of a devastating complication. Ann Surg 184:194-204, 1976.

5. Cooper JO: Trachea-innominate artery fistula: Successful management of three consecutive patients. Ann Thorac Surg 24:439-447, 1977.

6. Pearson FG, Goldberg M, DaSilva AJ: A prospective study of tracheal Injury complicating tracheostomy with a cuffed tube. Ann Otol Rhinol Laryngol 77:867-882, 1968. 7. Stiles PJ: Tracheal lesions after tracheostomy. Thorax 20:517-522, 1965.

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8. Mulder OS, Rubush Jl: Complications of tracheostomy: Relationship to long-term ventilatory assistance. } Trauma 9:389-402, 1969.

11. Kapur TR: late post-radiation changes in the larynx, pharynx, esophagus, and the trachea. } Laryngol 010/82:447-457, 1968.

9. Andrews MI, Pearson FG: Incidence and pathogenesis of tracheal injury following cuffed tube tracheostomy with assisted ventilation: Analysis of a two-year prospective study.

12 Phillips Tl, Fu KK: Acute and late effects of multimodal therapy on normal tissues. Cancer 40:489-494, 19n.

Ann Surg 173:249-263, 1971. 10. Conn J Jr, Tolis GA, Shields TW: Fatal hemorrhage following tracheostomy. 1M} 135:

13. Silen W, Speeker 0: Fatal hemorrhage from the innominate artery after tracheostomy.

TJ-29, 1969.

Ann Surg 162:1005-1012, 1965.

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Suggestions for monitoring patients during treatment with aminoglycoside antibiotics.

18S TRACHEOINNOMINATE ARTERY FISTULA AS A COMPLICATION OF RADIATION THERAPY DAVID REITER, DMD, MD BRUCE R. PICCONE PHltlP UnMAN, MD SHElDON A. USKER...
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