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Interact CardioVasc Thorac Surg 2007;6:484-489. doi:10.1510/icvts.2007.152280 © 2007 European Association of Cardio-Thoracic Surgery
Management and surgical resection for tracheobronchial tumors – institutional experience with 12 patients
a Department of General Thoracic Surgery, Toneyama National Hospital, Toneyama 5-1-1, Toyonaka City, Osaka 560-8552, Japan Received 16 January 2007; received in revised form 31 March 2007; accepted 23 April 2007
*Corresponding author. Tel.: +81-6-6853-2001; fax: +81-6-6850-1750.
We reviewed the records of 12 patients with primary tracheobronchial tumors and various clinical characteristics treated at our institution to investigate our overall management experience with disease. Over a 21-year period, we treated 1405 cases of primary pulmonary neoplasms, of which 12 (0.9%) patients had primary tracheobronchial tumors with eight different histological types, including three adenoid cystic carcinomas, two bronchial carcinoids, two papillomas, one squamous cell carcinoma, one mucous gland adenoma, one inflammatory pseudotumor, one schwannoma, and one mucoepidermoid carcinoma. Eleven of the patients had symptoms of airway obstruction and/or secondary infection or bleeding. A complete resection was performed in ten, which included a sleeve lobectomy in seven, sleeve pneumonectomy in one, tracheal resection in one, and left main stem resection without lung resection in one. Median survival following complete resection was 91 months. When possible, a complete resection provides the best potential benefits and symptomatic relief for patients with tracheo-endobronchial tumors. Further, various options related to tracheobronchoplasty including conservative resection can be applied for surgical intervention.
Key Words: Tracheobronchial tumors; Bronchoplasty; Clinical spectrum
Primary tracheobronchial tumors are relatively rare neoplasms found in the trachea, carina and endobronchial regions. Due to their rarity and variety of histogeneses as well as the fact that they do not appear on X-ray in most cases, their clinical characteristics and operative results have not been well established [1, 2]. These issues prompted us to review the published reports and our institutional experience, including characteristic case presentations. We found that a variety of histologic pictures of the disease entities have been reported, including benign, low, and high-grade malignancies [1, 3–5]. The clinical picture of tracheobronchial tumors usually divides them into two groups; those with airway obstruction that are treated as asthma and eventually become a life-threatening emergency, and those associated with a secondary pulmonary infection [1, 3, 5]. However, those symptoms are unspecific and insidious, due to the slow growth demonstrated by these tumors. As for the surgical interventions, a tracheo-bronchoplasty with or without lung resection [6, 7] is often employed for these tumors because of their nature. We retrospectively reviewed 12 cases of primary tracheobronchial tumors with eight different histological types treated at our institution, and herein describe some of the case presentations as well as characteristic clinical and radiologic features of this rare disease. Further, we discuss the diagnostic and therapeutic modalities of the disease.
From 1985 to 2005, we treated 1405 primary pulmonary or tracheo-endobronchial neoplasms (85 benign and 1320 malignant) at our hospital, including 12 (0.9%) patients with primary tracheobronchial tumors (four tracheal and eight endobronchial tumors). For the present analysis we excluded cases that underwent a tracheal resection for an invaded thyroid carcinoma or endobronchial metastasis via other primary sites, and those with locally advanced lung cancer that had invaded into the trachea or bronchus. There were two men and ten women, whose ages ranged from 15 to 85 years (mean 54.9 years), of whom five had a benign tumor and seven had a malignant tumor including six with low-grade malignancy. The last follow-up examination was in September 2006, and the mean follow-up period was 110 months. At the end of the study, nine patients were alive and three had died, two from tumor recurrence.
Patient demographic profiles are summarized in Table 1. Eight histological types of tumor were found in the 12 patients, including three adenoid cystic carcinomas, two bronchial carcinoids, two papillomas, one mucous gland adenoma, one squamous cell carcinoma, one inflammatory myofibroblastic tumor (IMT), one schwannoma, and one mucoepidermoid carcinoma. Eleven (91.7%) patients had symptoms of airway obstruction and/or secondary infection, while the other patient had no symptoms and the tumor was found incidentally at an annual check-up for concurrent brain meningioma. Radiologically, the maximum tumor diameter ranged from 1.0 to 6.5 cm (mean 3.3 cm).
Operative procedures for curative resection were performed for ten patients, which included seven sleeve lobectomies, one sleeve pneumonectomy, one tracheal resection and reconstruction, and one left main stem resection without lung resection. There was no postoperative morbidity in any of the 12 cases. Endoscopic laser ablation was attempted in an octogenarian patient with a papilloma (Case 2), who died of myocardial infarction without tumor recurrence. One patient (Case 12) with unresectable squamous cell carcinoma was treated by laser ablation, followed by radiotherapy. Two patients with adenoid cystic carcinoma and atypical carcinoids (Cases 6 and 11) died of recurrence of the tumors. The median survival was 91 months for ten patients who had a complete resection. Following are five case reports that illustrate some clinical important features of the various types of tracheobronchial tumors. 3.1.1. Case 1A 49-year-old female was admitted for close evaluation of a recent onset of wheezing that was not resolved by medication, and bronchofiberscopy revealed a protruding mass in the left main bronchus. Since the papilloma tumor was benign in nature, a bronchial resection without a lobectomy was performed to achieve curative but conservative resection. Histologically, the tumor was found to be a well-differentiated squamous cell papilloma with fibrous stroma (Fig. 1). However, relapse occurred at the proximal portion of the resected bronchus, which was endoscopically resected. Human papillomavirus-6b was detected in the resected recurrent papilloma specimen by a polymerase chain (PCR).
3.1.2. Case 3 A 71-year-old male was admitted complaining of a recent onset of strider and dyspnea. Chest X-ray revealed an atelectasis in the left upper lobe, and a bronchofiberscopy showed a well-circumscribed lobulated obstructing mass in left B4+5. Needle aspiration biopsy and cytology revealed a suspicious adenoma with mild atypism, with cytology results also showing that the cells were regular with basally situated distinct small nuclei, and cytoplasm had copious mucinous material (Fig. 2a). We could not exclude the possibility of lung cancer including low-grade malignancy, thus, a left upper sleeve lobectomy was performed for complete cure (Fig. 2b). A histological diagnosis of the resected specimen was consistent with a benign mucus gland adenoma without invasion to the bronchial cartilage [7]. Microscopically, the tumor was composed of a granular and tubulocystic arrangement with minimal pleomorphism.
3.1.3. Case 5 A 50-year-old female was admitted to our hospital with a recurrent hemoptysis. A chest computed tomogram (CT) revealed a mass of 3.5 cm in size in B7 (Fig. 3a) and a bronchofiberscopy confirmed a protruding mass with bleeding. During surgery, the tumor was diagnosed as a pulmonary sarcoma by frozen section histology, and a wedge sleeve lobectomy with a complete lymphadenectomy was performed. Histologically, the tumor was found to be an inflammatory myofibroblastic tumor (IMT) [8] without evidence of malignancy (Fig. 3b). This case was sub-categorized as a plasma cell granuloma with a similar polymorphic pattern which was confirmed by immunohistochemistry and positive finding for anaplastic lymphoma kinase (ALK) being positive.
3.1.4. Case 7 A 75-year-old female had been treated for bronchial asthma for three years, after which she was admitted for progressive exacerbation of strider and dyspnea, despite intensive medical therapy. Chest X-ray revealed normal, however, a mass was found in the trachea by chest CT and bronchoscopy (Fig. 4a,b). An emergency laser ablation followed by a circumstantial tracheal resection with a telescoping technique were then performed, after which a histological diagnosis of adenoid cystic carcinoma was obtained.
3.1.5. Case 9 A 15-year-old female was admitted for close evaluation of a recent onset of right lower lobe atelectasis associated with pulmonary infection. A chest CT revealed a protruding mass in the right intermedius bronchus (Fig. 5a). A bronchofiberscopy revealed a protruding mass, however, definite histological diagnosis was not obtained. At surgery, the right middle and lower lobes demonstrated chronic infection and the interlobar lymph nodes were swollen. A sleeve resection of the right middle and lower lobes was performed to achieve a curative surgical resection. The tumor was diagnosed as a mucoepidermoid carcinoma (Fig. 5b). The patient is doing well 34 months after surgery.
Clinically, a tracheobronchial tumor usually causes an airway obstruction and secondary pulmonary infection, though the characteristics are nonspecific [1–5]. In the current series, all patients with a detectable tumor in chest X-ray images were symptomatic except one. Delays in diagnosis of tracheal or endobronchial tumors commonly occur, because of negative chest X-ray findings and non-specific symptoms. A cross-sectional CT imaging was an important and complementary role for bronchoscopy [3]. In addition, modern tracheobronchial surgery procedures including not only resection and reconstruction of the airway, but also interventional bronchoscopy, are important for diagnosis and emergency treatment. Tracheobronchial tumors encompass a variety of benign, or low- and high-grade malignant characteristics. Thus, CT is useful to define the unsuspected lesions associated with tracheobronchial malignancies. Further, an emergent virtual bronchoscopy technique with the aid of 3-dimensional (3-D) reconstruction of high resolution helical CT images provides a unique opportunity to assess the extra- and intra-luminal pathologies of the tracheobronchial tumors. 4.1. Benign tracheobronchial tumors There are a variety of histologic features of this disease entity including benign, and low- and high-grade malignancies. However, since most physicians rarely experience these tumors due to their rarity, a precise preoperative diagnosis is difficult to obtain. Further, there is no unanimity of opinion regarding treatment. Solitary papillomas [9, 11], mucous gland adenomas [9], inflammatory myofibroblastic tumors (IMTs) [2, 8], schwannomas [2, 10, 11], leiomyomas [2, 3, 5], hamartomas [2], hemangiomas [2, 12] and chondromas [2, 12] have also been reported as rare benign tracheobronchial neoplasms. Recently, endoscopic resections have been applied for small benign lesions [2, 10, 12], which we also performed in Cases 1 and 2 for treatment of papillomas. In Case 1, a papillomavirus, which might have been related with the disease that was detected by a polymerase chain reaction (PCR) assay of the resected specimen, as has been previously reported [9]. Also, a schwannoma (neurogenic tumor), an extremely rare tracheobronchial tumor with an usually large size [11], was presented in the present Case 4 and mucous gland adenoma, which arose from the bronchial mucus glands and was essentially benign with minimal atypism [7], was seen in Case 3.Alternatively, IMTs are challenging lesions with respect to classification, differential diagnosis, and biologic potential [8], and are usually presented as a solitary parenchymal mass rather than an endobronchial lesion. Histologically, IMTs reveal spindle cell proliferation with a distinctive fibro-inflammatory and even pseudosarcomatous appearance [8], and have been described by various terms based on the variable cellular components, including plasma cell granuloma, and inflammatory pseudotumor. However, whether the pathogenesis is an inflammatory reactive lesion or neoplastic still remains controversial, though recent studies [8] have suggested that an IMT is a neoplasm. 4.2. Low-grade malignant tracheobronchial tumors In addition to squamous cell carcinomas of the tracheobronchial tree [1, 5], there are three other relatively uncommon tumors in this category, including carcinoid tumors, adenoid cystic carcinoma [1, 3, 13] and mucoepidermoid carcinomas [2, 6, 13], which are regarded as the three major subtypes of those tumors with low-grade malignancy. All six patients in the present series with these types of tumors underwent a tracheobronchoplasty as a curative resection. One patient with atypical carcinoids and one with adenoid cystic carcinoma died of distant metastasis despite adjuvant postoperative chemo-radiotherapy at 23 months and 118 months, respectively.An adenoid cystic carcinoma, most commonly encountered in the salivary glands, presents a gross endoscopic appearance that is indistinguishable from usual bronchogenic carcinomas. These tumors are generally regarded as a low-grade malignant potential [13, 14], however, they have a tendency for local recurrence and late development of distant metastasis, as seen in the present Cases 6 and 8. In another study, detailed histological examinations revealed extensive submucosal spreading that directly transgressed the bronchial wall and invaded into the pulmonary parenchyma [14], which were different from mucoepidermoid carcinomas [13, 15]. Nomori and colleagues [14] further categorized the adenoid cystic carcinomas into three grades according to the infiltration into the bronchial wall, and found that the grade was correlated with histology and prognosis. Mucoepidermoid carcinomas are regarded to be less common and have a lower level of malignancy than adenoid cystic carcinomas in terms of airway wall extension [13–15]. Five-year survival is 93–100% after complete resection [6, 13] compared to adenoid cystic carcinomas of 57–79% [1, 13, 14]. In our institutional experience, we have encountered only two cases with endobronchial carcinoids (one typical and one atypical), which is a lower percentage than in other reports [2–4]. We considered that this is probably due to the fact that peripheral endobronchial carcinoids are classified as lung cancer in the periphery and we did not analyze on the bronchial carcinoids in this series. Thus, our patients with endobronchial low-grade malignancy, including carcinoids, adenoid cystic carcinomas, and mucoepidermoid carcinomas obtained a survival benefit by undergoing a complete resection. Further, a conservative resection to avoid a pneumonectomy is warranted because of the low-grade malignancy. We concluded that, when possible, a complete resection provides the best potential benefit as well as symptomatic relief for patients with tracheobronchial tumors. Further, various options of tracheobronchoplasty procedures, including conservative resection, can be applied for that surgical intervention.
The authors wish to thank Dr. Osamu Kuwahara, Dr. Mitsunori Ohta, Dr. Keiji Inada, Dr. Hiroki Asada and Dr. Kiyohiro Fujiwara, former attending surgeons, for their contributions to this project, and Miss Yukari Hirai for her secretarial assistance.
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Abstract
1. Introduction
