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Pulmonary Artery Tumor Embolism in a Patient With Previous Fibroblastic Osteosarcoma

2013; Elsevier BV; Volume: 95; Issue: 6 Linguagem: Inglês

10.1016/j.athoracsur.2012.10.062

1552-6259

Silviu Buderi, Thomas Theologou, John R. Gosney, Michael Shackcloth,

Medical Imaging Techniques and Applications

A 48-year-old man was referred for left pulmonary metastasis and a left pulmonary artery embolus. The patient had T-cell acute lymphoblastic leukemia and fibroblastic osteosarcoma. A left pneumonectomy was performed successfully and the histologic report concluded that an embolic deposit of osteosarcoma was present. Pulmonary artery tumor embolism is a rare presentation in patients with previous fibroblastic osteosarcoma. It is important to suspect this diagnosis in a patient with cancer who presents with a pulmonary artery embolus. A 48-year-old man was referred for left pulmonary metastasis and a left pulmonary artery embolus. The patient had T-cell acute lymphoblastic leukemia and fibroblastic osteosarcoma. A left pneumonectomy was performed successfully and the histologic report concluded that an embolic deposit of osteosarcoma was present. Pulmonary artery tumor embolism is a rare presentation in patients with previous fibroblastic osteosarcoma. It is important to suspect this diagnosis in a patient with cancer who presents with a pulmonary artery embolus. Pulmonary artery tumor embolism has a prevalence of 10% in the patients with cancer. It is more common in patients with hematogenic neoplasms and is known to cause sudden death [1Chomette G. Auriol M. Sevestre C. Acar J. Metastatic pulmonary emboli: clinical types and incidence.Ann Med Interne (Paris). 1980; 131: 217-221PubMed Google Scholar]. The existence of acute lymphoblastic leukemia and osteosarcoma has been debated previously in the literature from the genetic susceptibility point of view but with no conclusive evidence of an association. However because of hematogenous spread, pulmonary metastasis is common in patients with osteosarcoma. We present a rare case of a patient with acute lymphoblastic leukemia and fibroblastic osteosarcoma, both treated successfully, who presented acutely with a pulmonary artery tumor embolism and pulmonary infarction. A 48-year-old man was referred for potential left pulmonary metastasis and left pulmonary artery embolus (Fig 1). His medical problems started in 1996 when he was diagnosed with T-cell acute lymphoblastic leukemia and underwent marrow transplantation after prolonged chemotherapy. In 2011, he began complaining of a painful bony mass in his left tibia, which was eventually diagnosed as fibroblastic osteosarcoma. The patient underwent a limb salvage surgical procedure after a course of neoadjuvant chemotherapy. There was a 4-month symptom-free interval before dyspnea developed exacerbated by exercise. His health deteriorated progressively to the point that he was severely dyspneic after only 1 flight of stairs. A computed tomographic scan revealed a left pulmonary mass and a left pulmonary artery embolus. Because of the size of the tumor (Fig 2), he required further neoadjuvant chemotherapy. After 4 cycles of chemotherapy with good clinical and radiologic response, he underwent a left pneumonectomy with no intraoperative or postoperative complications, and he was discharged on the fifth postoperative day. Examination of the resected specimen revealed complete occlusion of the left main pulmonary artery by an embolic deposit of osteosarcoma that had begun to organize and was adherent to the arterial wall (Fig 3). There was thrombosis of distal pulmonary arterial branches, and the pulmonary mass was revealed to be an organizing pulmonary infarct. No tumor was identified within the lung.Fig 3Low-power view of tumor embolus (green arrow) adherent to the wall of pulmonary artery (blue arrow) top right (hematoxylin and eosin, original magnifcation ×4).View Large Image Figure ViewerDownload (PPT) The patient was discharged on the fifth postoperative day without complications. During a postoperative follow-up at 6 weeks after the procedure, the patient presented an uneventful recovery period. Carcinomas of breast, stomach, colon, kidney, and prostate; choriocarcinoma; and hepatocellular carcinoma are the most common neoplasms giving rise to pulmonary metastases, including pulmonary artery tumor embolism [2Hadfield J.W. Sterling J.C. Wraight E.P. Multiple tumour emboli simulating a massive pulmonary embolus.Postgrad Med J. 1982; 58: 792-793Crossref PubMed Scopus (5) Google Scholar]. Pulmonary embolism into the pulmonary artery is rarely due to osteosarcoma, although occasional cases of embolizing chondrosarcoma have been reported. Ting and colleagues [3Ting P.T. Burrowes P.W. Gray R.R. Intravascular pulmonary metastases from sarcoma: appearance on computed tomography in 3 cases.Can Assoc Radiol J. 2005; 56: 214-218PubMed Google Scholar] emphasized the rarity of this event, presenting 3 cases of intravascular pulmonary metastasis, 2 caused by chondrosarcoma and 1 by osteosarcoma. Invasion of intratumoral blood vessels presumably results in embolism into the inferior vena cava, where the tumor reaches the pulmonary arterial circulation [4Hoekstra H.J. Pras B. Mooyaart E.L. Van Ginkel R. Molenaar W.M. Pelvic girdle chondrosarcoma and inferior vena cava thrombosis.Eur J Surg Oncol. 1997; 23: 577-579Abstract Full Text PDF PubMed Scopus (5) Google Scholar]. The clinical presentation is nonspecific—the patient presents with exertion dyspnea or cough or hemoptysis at rest—although a thorough history and high index of suspicion should suggest the cause, especially when unexplained hypoxia is present. Prolonged pulmonary hypertension with cor pulmonale is the most frequent chronic complication. Progressive dyspnea and right heart failure can induce sudden death. As stated by Winterbauer and colleagues [5Winterbauer R.H. Elfenbein I.B. Ball Jr, W.C. Incidence and clinical significance of tumor embolization to the lung.Am J Med. 1968; 45: 271-290Abstract Full Text PDF PubMed Scopus (214) Google Scholar] and Lee and associates [6Lee J.W. Lee K.H. Kim L. Yoon Y.H. Kim Y.J. Kim Y.J. Uncommon pulmonary metastasis presenting as pulmonary infarction with tumour emboli in two cases.J Med Imaging Radiat Oncol. 2012; 56: 192-194Crossref PubMed Scopus (1) Google Scholar], once cor pulmonale develops, survival is variable and has been reported as between 6 hours and a year. In terms of diagnosis, the computed tomographic pulmonary angiogram is the most efficient diagnostic tool. Karauzum and coworkers [7Miniero R. Barisone E. Vivenza C. et al.Acute lymphoblastic leukemia in a girl treated for osteosarcoma.Pediatr Hematol Oncol. 1995; 12: 185-188Crossref PubMed Scopus (9) Google Scholar] discussed the potential association between acute lymphoblastic leukemia osteosarcoma in an 8-year-old child with both conditions but were unable to determine any such link. In our patient, prompt diagnosis and good response to chemotherapy offered the possibility of surgical cure. It is of note that the tumor embolus occluded the pulmonary artery, causing distal thrombosis and infarction, but did not itself embolize into the lungs.