Asian Cardiovasc Thorac Ann 1999;7:144-146
© 1999 Asia Publishing EXchange Pte Ltd
Tracheal Adenoid Cystic Carcinoma Mimicking Bronchial Asthma
Yi
it Akçali, MD,
Fahri Ouzkaya, MD,
Cemal Kahraman, MD,
Leyla Hasdiraz, MD
Department of Thoracic and Cardiovascular Surgery
Erciyes University Medical Faculty
Kayseri, Turkey
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For reprint information contact: Yiit Akçali, MD Tel: 90 352 437 7683 Fax: 90 352 437 5285 email: yakcali{at}service.raksnet.com.tr Department of Thoracic and Cardiovascular Surgery, Erciyes University Medical Faculty, Kayseri 38039, Turkey.
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Abstract
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Adenoid cystic carcinomas are rare tracheal tumors that can mimic bronchial asthma. We describe the characteristics of 2 patients with adenoid cystic carcinoma who presented with symptoms suggestive of bronchial asthma of 2 years duration. Biopsy specimens revealed that the lesions were adenoid cystic carcinoma of the trachea. Both patients underwent collar incision and median sternotomy, the tumors were resected, and the tracheas were reconstructed primarily. Postoperative radiotherapy was given. Their postoperative clinical courses were uneventful and no local recurrences were seen at the 1-year follow-up.
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Introduction
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Tracheal tumors can be categorized as either primary (much less common than neoplasms of the lung or larynx) or secondary tumors that usually involve the trachea and carina by direct invasion from adjacent tissue such as the origin of the larynx, the thyroid, lung, or esophagus. Tracheal adenoid cystic carcinoma (ACC) is a rare malignancy that may cause signs and symptoms that mimic common upper airway diseases.1,2 Primary tracheal tumors are best treated by resection when primary reconstruction can be safely accomplished. Resection should be followed by postoperative irradiation. Early diagnosis of ACC is important because appropriate surgical treatment results in an excellent prognosis.2
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Case Reports
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Case 1
A 51-year-old man was admitted to our department because of persistent signs of status asthmaticus for approximately 2 years, which did not respond to bronchodilator therapy. He had no history of asthma. Tracheoscopy showed a sessile tumor obstructing two-thirds of the lumen, 6 cm above the carina. A biopsy showed the tumor to be an ACC. It was excised via a collar incision and median sternotomy, the right hilum was released, and the trachea was reconstructed by end-to-end anastomosis with interrupted sutures. His chin was sutured to the skin of the manubrium to prevent neck extension during the first postoperative week. Postoperative radiotherapy was given and the postoperative clinical course was uneventful. There was no local recurrence detected at follow-up 1 year later.
Case 2
A 45-year-old woman was transferred to our department with respiratory distress attributed to an asthma-like attack. She reported symptoms of 2 years duration suggestive of bronchial asthma, which did not respond to therapy with ß-2 agonists. Magnetic resonance imaging (Figure 1
) and computed tomography showed a tracheal mass. Tracheobronchoscopy revealed a protruding hemispheric lesion on the membranous portion of the trachea 3 cm below the glottis. The biopsy specimen indicated that the lesion was an ACC of the trachea. Via collar incision and median sternotomy, a 6-cm length (10 rings) of the trachea was resected (Figure 2), right hilar pulmonary release was performed, and the trachea was reconstructed by end-to-end anastomosis with interrupted sutures. Her chin was sutured as described in the first patient. Postoperative radiation therapy was given. Her postoperative clinical course was uneventful without serious anastomotic complications. No local recurrence was seen 1 year after the operation.
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Figure 1. Magnetic resonance image demonstrating a solid tumor mass in the trachea 32 mm below the glottis. The tracheal wall has been invaded posteriorly and bilaterally by the mass and the pretracheal fat tissue has been infiltrated. The esophagus has been displaced by the tumor and the tracheoesophageal planes have been obliterated.
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Discussion
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ACC was previously called "cylindroma" and its gross appearance is benign as the overlying tracheal mucosa is frequently intact and the progression of such tumors is exceedingly slow. However, ACC is nearly always found to extend beyond the visible and palpable confines of the gross lesion at the time of surgery. The posterior wall is the most frequently involved site.3 Frozen-section evaluation of resection margins is crucial to successful excision. Metastasis to regional lymph nodes is reported in approximately 10% of patients.3 Hematogenous metastasis occurs most commonly in the lungs but there is occasional spread to brain and bone. Even in untreated cases, the natural history of ACC is often that of a slow and insidious progression.4
Clinical manifestations of ACC may result from upper airway obstruction, mucosal irritation and ulceration, direct invasion and involvement of contiguous structures, or as the result of distant metastasis. Many patients with ACC are diagnosed as having asthma or chronic bronchitis for long periods prior to the correct diagnosis because upper airway obstruction is characterized by dyspnea, stridor, wheezing, and cough. Pulmonary function tests may alert the physician to the possibility of tracheal obstruction. Flow-volume loops have a typical flattening of both inspiratory and expiratory curves associated with a fixed obstruction in cases of tracheal tumor.
Tracheobronchoscopy is essential and provides the simplest and most reliable approach for tumor diagnosis. Surgical excision with primary reconstruction is the best treatment modality for ACC. Pearson and colleagues5 advocated adjuvant radiation therapy for all patients with tracheal ACC because of its high radiosensitivity. All patients who undergo resections of such tumors should receive full-dose mediastinal irradiation postoperatively, regardless of whether the margins or the adjacent lymph nodes are negative.5,6 Long-term survival is excellent in patients with ACC. Among patients with tracheal cancers, survival is significantly longer for those with complete resections than for those with incomplete resections. On the other hand, the presence of positive lymph nodes does not seem to decrease survival. It is important to note that all patients with ACC reported by different authors received adjuvant radiothrapy.6–8 Lethal complications include anastomotic dehiscence, pneumonia and respiratory failure, pulmonary embolism, and tracheal innominate artery fistula.4 Tracheal ACC should be considered when patients with bronchial asthma do not respond to bronchodilator treatment.
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References
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Abstract
Introduction
