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Journal of Clinical Oncology, Vol 25, No 34 (December 1), 2007: pp. 5521-5523 © 2007 American Society of Clinical Oncology. DOI: 10.1200/JCO.2007.13.7273
Unresectable Adenoid Cystic Carcinoma of the Trachea Treated With ChemoradiationDepartments of Radiation Oncology, Medicine, and Surgery, Brigham and Women's Hospital and Harvard Medical School, Boston, MA
Medical Oncology, Departments of Radiation Oncology, Medicine, and Surgery, Brigham and Women's Hospital and Harvard Medical School, Boston, MA
Pulmonary Medicine, Departments of Radiation Oncology, Medicine, and Surgery, Brigham and Women's Hospital and Harvard Medical School, Boston, MA
Thoracic Surgery, Dana-Farber Cancer Institute; Departments of Radiation Oncology, Medicine, and Surgery, Brigham and Women's Hospital and Harvard Medical School, Boston, MA A 30-year-old female developed dyspnea with exertion during the second trimester of her first pregnancy. After a normal delivery she continued to have shortness of breath that went unexplained. A diagnosis of asthma was made and a trial of bronchodilators was begun. Dyspnea continued without improvement and a dry cough began. Chest computed tomography (CT) and magnetic resonance imaging (MRI) scans were performed that showed a soft tissue mass surrounding the trachea extending from the level of the thoracic inlet to the carina. Chest imaging, including both CT and MRI scans, demonstrated intramural tumor extending from 6 cm below the vocal cords to the orifice of the right upper lobe bronchus. Contrast-enhanced CT scans demonstrated no paratracheal adenopathy or pulmonary parenchymal disease (Fig 1A). T2-weighted MRI sequences with gadolinium administration were slightly more sensitive than CT scanning in demonstrating the submucosal extent of the tumor (Fig 1B).
The patient underwent a bronchoscopy that showed mural irregularities extending from six rings below the vocal cords into the right mainstem bronchus. The circumfererential submucosal masses protruded into tracheal lumen and demonstrated striking neovascularity. Figure 2 shows the visible tumor in the distal trachea. Planar reconstruction of the CT scan images demonstrated tumor in the distal trachea (Fig 2A). Bronchoscopic examination of the tumor demonstrated hyperemia and submucosal tumor (Figs 2B and C).
Bronchoscopic biopsies of the mural lesions showed adenoid cystic carcinoma infiltrating respiratory mucosa. A positron emission tomography/CT scan was performed that showed no significant avidity in the mediastinum; no lung metastases were detected. Extended tracheal resection was considered. The primary contraindication to surgical resection was the extent of the tumor from the mid-carina into the right upper lobe bronchus. As a result, the patient was seen by a multidisciplinary team of medical and radiation oncologists and the decision was made to treat the patient with definitive chemoradiotherapy. The patient received 66 Gy of radiotherapy to the primary tumor volume. Figure 3 shows the radiation treatment plan. A coronal dose distribution of the radiation plan shows the extent of the treatment (Fig 3A). An axial figure shows the dose distribution of the radiation treatment plan (Fig 3B). The plan uses five fields (anterior, posterior, right lateral, right anterior oblique, and left anterior oblique). Pseudocoloring shows the high dose (shown in red) and lower doses (shown in green and blue, respectively).
The radiation therapy was administered with concurrent weekly carboplatin (area under the time-concentration curve 2) and paclitaxel 50 mg/m2 for 6 weeks. During the therapy, the patient developed mild esophagitis and fatigue, as expected, but was able to tolerate a modified diet throughout therapy. At the end of treatment, she noted that her exercise tolerance and shortness of breath were improved dramatically. A repeat CT scan performed 4 months after therapy showed a complete response to the combined-modality treatment. A chest CT scan of the patient 3 months after chemoradiotherapy therapy showed no residual tracheal tumor (Fig 4, arrow). The CT scan was interpreted as normal by the chest radiology consultant. Bronchoscopies performed 3 and 6 months after therapy showed normal mucosa. Surveillance biopsies showed no evidence of malignancy.
Adenoid cystic carcinoma (ACC) of the trachea is a relatively rare occurrence. Tracheal tumors as a whole represent 0.2% of respiratory malignancies and most of these are squamous cell carcinoma. Only 10% of these cases are adenoid cystic carcinoma.1 The primary management of tracheal ACC is surgical resection. Gaissert et al2 has published the largest series of tracheal ACCs, with 135 occurrences in patients at the Massachusetts General Hospital during 40 years; 71% of these patients were treated with resection and showed with a 52% 5-year overall survival. Radiotherapy was administered in 70% of the patients postoperatively. In the unresectable ACC patients, radiotherapy alone was recommended, with a overall survival of 30%.2 Unresectable ACC presents an increasingly difficult problem to control, given that the tumors often traverse long regions throughout the thorax. Primary radiotherapy has had mixed results, with local control varying between 20% and 70% (with some improvements with radiotherapy doses > 60 Gy).3,4 The use of concurrent chemotherapy combined with radiotherapy, although used commonly in other bronchogenic carcinomas, has been reported sparingly in ACC. Two limited reports have been published using combined therapy with good results.5,6 The combination of carboplatin/paclitaxel and radiotherapy has been effective in unresectable head and neck ACC with good results,7 and we are encouraged by the results of this patient and three others treated at Dana-Farber Cancer Institute/Brigham and Women's Hospital (Boston, MA) with a similar approach. ACC is a disease that combines locoregional treatment challenges with the risk of late pulmonary metastases. In patients with unresectable disease, the short-term effectiveness of combined chemoradiotherapy suggests that a curative approach is justifiable in tracheal ACC. AUTHORS' DISCLOSURES OF POTENTIAL CONFLICTS OF INTEREST The author(s) indicated no potential conflicts of interest.
REFERENCES 1. Gaissert HA, Mark EJ: Tracheobronchial gland tumors. Cancer Control 13:286-294, 2006[Medline] 2. Gaissert HA, Grillo HC, Shadmehr MB, et al: Long-term survival after resection of primary adenoid cystic and squamous cell carcinoma of the trachea and carina. Ann Thorac Surg 78:1889-1896, 2004; discussion 1896-1897 3. Fields JN, Rigaud G, Emami BN: Primary tumors of the trachea: Results of radiation therapy. Cancer 63:2429-2433, 1989[CrossRef][Medline] 4. Cheung AY: Radiotherapy for primary carcinoma of the trachea. Radiother Oncol 14:279-285, 1989[CrossRef][Medline] 5. Sasiaja M, Funa N, Kamata M, et al: Unresectable adenoid cystic carcinoma of the trachea treated with chemoradiotherapy. Clin Oncol (R Coll Radiol) 12:272, 2000 6. Videtic GM, Campbell C, Vincent MD: Primary chemoradiation as definitive treatment for unresectable cancer of the trachea. Can Respir J 10:143-144, 2003[Medline] 7. Haddad RI, Posner MR, Busse PM, et al: Chemoradiotherapy for adenoid cystic carcinoma: Preliminary results of an organ sparing approach. Am J Clin Oncol 29:153-157, 2006[CrossRef][Medline]
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Copyright © 2007 by the American Society of Clinical Oncology, Online ISSN: 1527-7755. Print ISSN: 0732-183X
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