Physical examination revealed a hard lump in the right breast, 10×10 mm size, not attached to underlying structures. Contralateral breast also had a 15 mm size lump. The right lump had been detected previously during a gynecologic examination several months earlier and was felt to be a fibroadenoma and close follow-up was suggested. Both lesions were excised and the histopathologic examination disclosed synovial sarcoma in the left mammary gland and confirmed fibroadenoma on the right side. Immunochemistry of the lung and left breast lesion were identical. A whole body positron emission tomography scan was performed and did not show any other site of metastatic lesion.
Metastatic tumors to the tracheobronchial tree are unusual, and occur in 2% of patients with solid tumors.1 The prevalence depends in fact on how the metastases are defined. If invasion of tracheobronchial structures by parenchymal or lymph node mass is included, the prevalence rate is higher. However, if they are defined as only direct metastases in the tracheobronchial tree from extrapulmonary lesions, the prevalence rate is much lower.8 The frequently associated primary tumors include those of the kidney, pancreas, colon, ovary, and thyroid.8 Even when the clinical and roentgenographic features of endobronchial metastasis and bronchogenic carcinoma are usually indistinguishable, in the majority of cases the primary tumor site is clinically apparent.1,9
Mammary carcinoma is likely to metastasize relatively early to the regional lymph nodes and thereafter primarily to the lungs, bone marrow, liver, and bone. Although pulmonary metastasis with invasion of a bronchus is reported, primary metastasis to an airway is distinctly rare.10 Whatever the histologic type, endobronchial metastasis tend to occur at a significant interval from the diagnosis of the primary tumor, indicating a relatively slow disease progression. A reported mean interval from the diagnosis of the primary carcinoma to the diagnosis of primary tracheobronchial tree involvement is between 59 months and 5.4 years9,11; however, the shortest reported time was 0 months in a patient with uterus carcinoma. In contrast to lung parenchymal metastases, the significantly lower number of reported tracheobronchial metastases is thought to be secondary to a difference in blood supply. The lung parenchyma is supplied by the pulmonary artery, through which all systemic blood passes; whereas, the trachea and bronchi are supplied by the bronchial artery receiving only a small portion of the systemic circulation. Thus, hematogenous spread is more likely to occur in the lung parenchyma.12
Synovial sarcoma is rare. It accounts for between 5% and 10% of the approximately 10,000 new soft tissue sarcomas reported each year.13 Most of the synovial sarcomas occur in the limbs. Primary pulmonary sarcoma has been described, but it is exceptional.14 In contrast, primary sarcoma occurring in breast is extremely rare and comprises 0.5% to 1% of all breast neoplasm. Only a few hundred cases of sarcomas other than cystosarcoma have been reported.15 In one of the few series of primary breast sarcomas, no single synovial sarcoma was observed in 42 cases.16 Because of the extremely unusual occurrence of this sarcoma in the breast, the possibility of a dual metastasis (lung and breast) or a primary lung sarcoma metastasizing in the breast was considered. The existence of a primary tumor in a more usual location seems to have been discarded by a negative positron emission tomography scan, which has shown to be extremely sensitive to detect sarcoma lesions.17 Extramammary malignancies metastasizing to the breast are also very rare and constitute approximately 2% of all breast tumors, but at least 1 case of synovial sarcoma with breast metastasis has been described.18
Our patient showed a combination of situations, which were each extremely unusual: a breast synovial sarcoma, an endobronchial metastasis of a soft-tissue sarcoma, and metastatic endobronchial disease as a first symptom of a malignant tumor. This exceptional scenario does not allow to accurately predict the survival expectancy. Unfortunately each one of these situations has a poor prognosis by itself. Primary sarcomas of the breast are aggressive tumors and the 3-year disease-free survival is approximately 39%.15 As only the margin of first surgery was found to be a significant predictor of survival, all attempts should be made to achieve a negative margin. In contrast, the presence of endobronchial metastasis also carries a short life expectancy. Survival after the diagnosis of endobronchial metastasis is poor with reported mean survival times between 12 and 15 months.9,11,19 However, as some studies have reported longer-term survival (21 to 32 mo), aggressive treatment should be considered.8,20
Clinical and epidemiologic features must be considered in the presence of an endobronchial lesion. Despite the unusual occurrence of EBM, primary lung cancer in a nonsmoking young woman is also very unusual.
Recently, molecular methods have been used for identification of specific mutations using a reverse transcription-polymerase chain reaction. These methods could be helpful to distinguish between pulmonary or extrapulmonary sarcomas, and to assess relationship with poor prognosis. However, it is still not available for clinical purposes.21
EBM are usually associated with a previously known primary tumor. In patients with endobronchial tumors, in contrast, metastatic soft-tissue sarcoma should be included in the differential diagnosis.
The authors thank Ms Paula De Bonis for her technical and secretarial assistance in preparing this manuscript.
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Keywords:© 2008 Lippincott Williams & Wilkins, Inc.
atelectasis; endobronchial electrosurgery; endobronchial metastases; interventional bronchoscopy; synovial sarcoma