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Ann Thorac Surg 2001;72:2111-2113
© 2001 The Society of Thoracic Surgeons
a Service de Pneumologie, Centre Cardio-Pneumologique, Hôpital Pontchaillou-CHU, Rennes, France
b Service de Chirurgie Thoracique et Cardio-Vasculaire, Centre Cardio-Pneumologique, Hôpital Pontchaillou-CHU, Rennes, France
c Service d’Oncologie Médicale, Centre Eugène Marquis, Rennes, France
Accepted for publication March 1, 2001.
* Address reprint requests to Dr Delaval, Service de Pneumologie, Centre Cardio-Pneumologique, Hôpital Pontchaillou-CHU, 35033 Rennes Cedex, France
e-mail: philippe.delaval{at}chu-rennes.fr
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Abstract |
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Introduction |
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We report the case of a man (born in 1962) with recurrent pneumothorax. Osteoblastic osteosarcoma of the inferior extremity of the left femur was diagnosed in 1989 and treated by neoadjuvant chemotherapy, followed by a limb-sparing surgical procedure with insertion of a knee prosthesis and then adjuvant chemotherapy. In the initial extension assessment, chest tomography revealed no metastasis. Complete remission was obtained in September 1989.
In January 1993, the patient was hospitalized with a completely spontaneous right pneumothorax, which required placement of a chest tube. A chest computed tomographic scan revealed a thin-walled cavity with a small calcified nodule in its inner edge, in the upper right axillary region (Fig 1). The possibility of a secondary location was considered and we decided to perform computed tomography 2 months later to evaluate the progression of the cavitary lesion. The patient did not show up for this examination.
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A chest computed tomographic scan showed the same cystic lesion, scarcely increased. The calcified nodule was unchanged. However, the wall of the cavitary lesion appeared thickened. There was perilobular and pleural thickening close to the lesion, which remained unique.
The nodule was completely excised surgically by means of thoracotomy. The nodule showed histologic features of metastasis of osteosarcoma with pleural extension. Two neoplastic arterial emboli were noted.
Two months after the operation, a chest computed tomographic scan revealed a stapler’s line, surrounded by linear thickenings and ground-glass areas, with pleural thickening occupying the space left by excision. This was considered to be secondary to the surgical procedure and had partly disappeared on a computed tomographic scan performed 14 months later (Fig 2).
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Comment |
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Isolated cases and small series of pneumothorax associated with lung metastasis have been described in children and young adults, with frequent occurrence of bone sarcoma. Chemotherapy of osteosarcoma leads to tumor necrosis thereby increasing the risk of pneumothorax, which occurs 1 to 8 days after initiation of treatment. In the future pneumothorax may be a more frequent complication of chemosensitive cancers [1].
Osteosarcoma is the most frequent tumor among children and young adults. Prognosis has improved since introduction of neoadjuvant and adjuvant chemotherapies before and after surgical excision. A high-dose methotrexate regimen is the standard treatment. Recovery is achieved in 70% of nonmetastatic forms [2].
The lung is the first site of dissemination of osteosarcoma [2]. At diagnosis only 10% of patients have lung metastasis, compared with 45% to 70% on autopsy [3]. A 50% relapse rate was noted in a retrospective study [2] of 56 patients treated with neoadjuvant chemotherapy and surgical excision, with or without adjuvant chemotherapy depending on the grade of malignancy. Lung metastasis occurred in 79% of cases without other evidence of dissemination. The median time of relapse was 15 months.
Lung metastasis usually appears early. In a retrospective study [4], metastatic relapse was seen in 84% of 279 patients with high-grade localized or locally widened osteosarcoma, without metastatic forms from the outset. The median time of occurrence was 11 months, with most metastases appearing at the end of chemotherapy. In one case study, a solitary metastatic pulmonary nodule appeared 67 months after completion of chemotherapy [4].
Lung metastasis of osteosarcoma is usually multiple and bilateral, but is unique in 35% of cases: 49% are parenchymatous and 51% are subpleural, 5% of the latter being complicated by pneumothorax [3]. Lung metastases of osteosarcoma are often calcified, more rarely bullous. These bullous metastatic lesions can progress to full nodules.
Chest computed tomography appears to be the best technique for detection and quantification of secondary pulmonary locations, particularly in subpleural areas. In a retrospective study of 32 patients, chest x-ray radiography and chest computed tomography were concurrently performed 57 times: the sensitivity of detection of lung metastasis was 57% for chest x-ray radiography and 88% for chest computed tomography [5]. Thick-section spiral computed tomography detects small nodules (< 5 mm), whereas nodules more than 20 mm in diameter could escape detection with conventional computed tomography because of the quality of inspiration.
Surgical excision of lung metastasis of osteosarcoma is the most efficient treatment. Ferrari and colleagues [6] showed that in the absence of surgical excision all patients died 40 months after relapse, whereas after surgical treatment the 10-year survival rate was 30%. Another study [2] indicated 4-year and 5-year survival rates of 16% and 36%, respectively, after surgical excision. The extent of necrosis does not seem to be a prognostic factor in lung metastasis.
Two conditions must be met if surgical resection of lung metastasis of osteosarcoma is to be considered: efficient treatment of the primary tumor and the extrathoracic metastasis, and the feasibility of complete resection compatible with pulmonary function. Surgical treatment is not indicated in the case of rapid metastatic growth, or if metastasis occurs soon after efficient treatment of the primary tumor [2].
Prognostic factors after surgical resection of lung metastasis of osteosarcoma have been studied by various investigators. Using multivariate analysis, gender is a factor, as for the same number of metastases (< 5), median survival was 280 days for women versus 560 days for men. Survival is longer when fewer nodules are resected, with a threshold of four to six nodules depending on the study [2]. The number of pulmonary lobes involved has also been considered significant.
In conclusion, our case presents several particularities: (1) Only 1% of cases of pneumothorax are due to lung metastasis; (2) A chest computed tomographic image of a bullous lesion with calcification is suggestive of metastasis in an osteosarcoma-bearing patient; (3) The time to relapse is 48 months after initial diagnosis and 40 months after the end of chemotherapy; (4) A favorable outcome as the patient is still in complete remission 7 years after surgical treatment of this single metastasis.
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This article has been cited by other articles:
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  J-T Lin, C-C Yen, W-S Wang, T-J Chiou, J-H Liu, H-T Wu, and P-M Chen Unusual peritoneal spreading by metastatic osteosarcoma of the tibia Br. J. Radiol., May 1, 2003; 76(905): 337 - 338. [Abstract] [Full Text] [PDF]
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