Lung (2015) 193:151–153 DOI 10.1007/s00408-014-9659-5
CASE REPORT
An Unusual Cause of Tree-in-Bud Pattern: Pulmonary Intravascular Tumor Embolism Caused by Chondrosarcoma Lilia Silva Peixoto • Paulo Marcos Valiante • Rosana Souza Rodrigues • Miriam Menna Barreto Gla´ucia Zanetti • Edson Marchiori
•
Received: 21 August 2014 / Accepted: 11 October 2014 / Published online: 16 October 2014 Ó Springer Science+Business Media New York 2014
Abstract We report the case of a 45-year-old man who initially presented with chondrosarcoma of the left femur that was treated surgically. Follow-up chest computed tomography (CT) performed 3 years later showed multiple small nodules with a tree-in-bud branching pattern and larger elongated opacities with beaded contours. These findings raised the suspicion of intravascular tumor embolism. Pulmonary CT angiography demonstrated intravascular thrombosis and dilated and beaded peripheral pulmonary arteries. The tumoral origin of the thrombus was confirmed by lung biopsy.
pattern and elongated opacities with beaded contours, raising the suspicion of pulmonary tumor embolism (Fig. 1). Pulmonary CT angiography demonstrated dilated and beaded peripheral pulmonary arteries and intravascular thrombus (Fig. 2). The patient underwent open lung biopsy. Histopathologic analysis demonstrated extensive metastatic intravascular tumor emboli (Fig. 3). The patient is undergoing maintenance chemotherapy and remains asymptomatic.
Discussion Keywords Tree-in-bud pattern Tumor embolism Chondrosarcoma metastasis Pulmonary diseases Computed tomography
Case Report A 45-year-old man presented with left knee pain caused by a tumor on the distal femur. A biopsy revealed grade III chondrosarcoma, and the tumor was surgically resected. The patient remained asymptomatic. Follow-up chest computed tomography (CT) performed 3 years later revealed multiple centrilobular nodules with a tree-in-bud
L. S. Peixoto P. M. Valiante R. S. Rodrigues M. M. Barreto G. Zanetti E. Marchiori (&) Federal University of Rio de Janeiro, Rua Thomaz Cameron, 438. Valparaiso. CEP 25685.120. Petro´polis, Rio De Janeiro, Brazil e-mail: [email protected] R. S. Rodrigues D’Or Institute for Research and Education, Rio De Janeiro, Brazil
Pulmonary tumor embolism refers to the spread of neoplastic cells throughout blood or lymphatic vessels [1, 2]. This entity is rare in blood vessels. Macroscopic and microscopic tumor emboli are often diagnosed in patients with previous malignancy [3]. This phenomenon occurs most frequently in patients with lung, gastrointestinal, liver, breast, and uterine neoplasia [4]. Few cases of chondrosarcoma causing tumor embolism have been reported, and the diagnosis was delayed in most instances [5–7]. Pulmonary intravascular tumor embolism is reported in 2.6–25 % of cancer cases. The actual frequency of this entity is difficult to estimate due to differences in methodologies and definitions. It may be more prevalent than reported because the diagnosis depends on histopathologic analysis, which is not performed in all cases of cancer [8]. Pulmonary tumor embolism is usually asymptomatic and underdiagnosed, and is often identified only on autopsy [4]. Our patient presented with no respiratory complaint, and the pulmonary nodules were depicted on routine imaging examinations. However, some patients present with dyspnea and (more rarely) cyanosis, cough, pleuritic
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Fig. 1 a Chest CT image obtained in the pulmonary window setting showing elongated opacities with beaded appearance (arrows) in the right lower lobe of the lung. b Note also the presence of multiple small nodules with the tree-in-bud pattern (arrows)
chest pain, hemoptysis, and/or hematic sputum. Rarely, as tumor cells occupy the pulmonary arteries and arterioles, subacute cor pulmonale may manifest as progressive dyspnea, which can lead to death [8]. Exceptionally, large tumoral emboli spread from a primary tumoral mass and obstruct main pulmonary arterial vessels, creating a clinical picture indistinguishable from massive pulmonary thromboembolism [4]. Thus, the possibility of tumor embolism should be considered when a patient with known malignancy develops dyspnea. The presentation of tumor embolism on chest CT ranges from the tree-in-bud pattern to the presence of an extensive thrombus occupying the right atrium and pulmonary artery trunk associated with areas of pulmonary infarction [8]. The tree-in-bud pattern, created by centrilobular branching structures, reflects a spectrum of endo- and peribronchiolar disorders, including mucoid impaction, inflammation, and fibrosis [9]. Originally reported in cases of endobronchial spread of Mycobacterium tuberculosis, this pattern is now recognized as a CT manifestation of many diverse entities,
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Fig. 2 a Angio-CT of the pulmonary arteries showed that the elongated opacities represented an extensive thrombus occupying the pulmonary vessel lumen. b Thrombi were also detected in the left lower lobe of the lung
including tumoral embolism [10]. As our patient had a history of treated tuberculosis, the finding of the tree-inbud pattern raised doubt about the possibility of new infection. The finding of elongated structures with softtissue density and beaded contours proximal to the centrilobular nodules raised the suspicion of a vascular origin of this pattern. CT angiography showed that those elongated structures corresponded to thrombosed vessels proximal to the tree-in-bud pattern. Histopathologic analysis confirmed the tumoral origin of the thrombus. Although pulmonary intravascular tumor embolism is not a common entity, this possibility should be suspected in patients with known malignancy presenting with the treein-bud pattern and angiotomographic positivity for pulmonary embolism. Cor pulmonale and, less often, massive tumor embolism with acute cardiorespiratory failure may occur in these cases. Recognition of the occurrence of tumor embolism is crucial in determining the most appropriate therapeutic approach for each patient.
Lung (2015) 193:151–153
153 Conflict of interest
The authors have no conflict of interest.
References 1. Kvale PA (1991) The cancer patient with dyspnea: unusual cause? (editorial). Mayo Clin Proc 66:215–218 2. Williams & Wilkins (1990). Stedman’s Medical Dictionary. 25th edn. Williams & Wilkins, Baltimore, p. 501 3. Jakel J, Ramaswamy A, Kohler U, Barth PJ (2006) Massive pulmonary tumor microembolism from a hepatocellular carcinoma. Pathol-Res Pract 202(5):395–399 4. Martos Pe´rez F, Seara Valero R, Weil Lara B, Contreras J, Domı´nguez Pico´n F (1997) Massive tumorous pulmonary embolism in hepatocarcinoma. An Med Interna 14(12):627–629 5. Leung DY, Seah PW, Lee LC, Cranney GB, Walsh WF (1993) Embolic chondrosarcoma: an unusual cause of pulmonary embolism. Am Heart J 126(3 Pt 1):732–734 6. Alirhayim Z (2014) Chondrosarcoma Presenting as a Fatal Pulmonary Embolism (abstract). J Hosp Med 9(2):309 7. Chandrasekharan R, Bhagavaldas MC, Mathew AJ (2011) Chondrosarcoma presenting as dyspnea in a 19-year-old man: a case report. J Med Case Rep. 5:150. doi:10.1186/1752-1947-5150 8. Soares FA (1991) Morphological and clinical aspects of the vascular involvement of the pulmonary vessels by tumor emboli. J de Pneumol 17(3):127–140 9. Hansell DM, Bankier AA, MacMahon H, McLoud TC, Mu¨ller NL, Remy J (2008) Fleischner Society: glossary of terms for thoracic imaging. Radiology 246:697–722 10. Rossi SE, Franquet T, Volpacchio M, Gime´nez A, Aguilar G (2005) Tree-in-bud pattern at thin-section CT of the lungs: radiologic-pathologic overview. RadioGraphics 25:789–801
Fig. 3 a Photomicrograph showing occupation of the vascular lumen by an intravascular node composed of neoplastic cells (original magnification 1009; hematoxylin and eosin stain). b Another intravascular node comprising neoplastic cells with chondroid differentiation, characterized by stellate nuclei and abundant hyaline matrix. The remainder of the parenchyma demonstrates congestion, necrosis, and hemorrhage (original magnification 1009; hematoxylin and eosin stain)
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CASE REPORT
An Unusual Cause of Tree-in-Bud Pattern: Pulmonary Intravascular Tumor Embolism Caused by Chondrosarcoma Lilia Silva Peixoto • Paulo Marcos Valiante • Rosana Souza Rodrigues • Miriam Menna Barreto Gla´ucia Zanetti • Edson Marchiori
•
Received: 21 August 2014 / Accepted: 11 October 2014 / Published online: 16 October 2014 Ó Springer Science+Business Media New York 2014
Abstract We report the case of a 45-year-old man who initially presented with chondrosarcoma of the left femur that was treated surgically. Follow-up chest computed tomography (CT) performed 3 years later showed multiple small nodules with a tree-in-bud branching pattern and larger elongated opacities with beaded contours. These findings raised the suspicion of intravascular tumor embolism. Pulmonary CT angiography demonstrated intravascular thrombosis and dilated and beaded peripheral pulmonary arteries. The tumoral origin of the thrombus was confirmed by lung biopsy.
pattern and elongated opacities with beaded contours, raising the suspicion of pulmonary tumor embolism (Fig. 1). Pulmonary CT angiography demonstrated dilated and beaded peripheral pulmonary arteries and intravascular thrombus (Fig. 2). The patient underwent open lung biopsy. Histopathologic analysis demonstrated extensive metastatic intravascular tumor emboli (Fig. 3). The patient is undergoing maintenance chemotherapy and remains asymptomatic.
Discussion Keywords Tree-in-bud pattern Tumor embolism Chondrosarcoma metastasis Pulmonary diseases Computed tomography
Case Report A 45-year-old man presented with left knee pain caused by a tumor on the distal femur. A biopsy revealed grade III chondrosarcoma, and the tumor was surgically resected. The patient remained asymptomatic. Follow-up chest computed tomography (CT) performed 3 years later revealed multiple centrilobular nodules with a tree-in-bud
L. S. Peixoto P. M. Valiante R. S. Rodrigues M. M. Barreto G. Zanetti E. Marchiori (&) Federal University of Rio de Janeiro, Rua Thomaz Cameron, 438. Valparaiso. CEP 25685.120. Petro´polis, Rio De Janeiro, Brazil e-mail: [email protected] R. S. Rodrigues D’Or Institute for Research and Education, Rio De Janeiro, Brazil
Pulmonary tumor embolism refers to the spread of neoplastic cells throughout blood or lymphatic vessels [1, 2]. This entity is rare in blood vessels. Macroscopic and microscopic tumor emboli are often diagnosed in patients with previous malignancy [3]. This phenomenon occurs most frequently in patients with lung, gastrointestinal, liver, breast, and uterine neoplasia [4]. Few cases of chondrosarcoma causing tumor embolism have been reported, and the diagnosis was delayed in most instances [5–7]. Pulmonary intravascular tumor embolism is reported in 2.6–25 % of cancer cases. The actual frequency of this entity is difficult to estimate due to differences in methodologies and definitions. It may be more prevalent than reported because the diagnosis depends on histopathologic analysis, which is not performed in all cases of cancer [8]. Pulmonary tumor embolism is usually asymptomatic and underdiagnosed, and is often identified only on autopsy [4]. Our patient presented with no respiratory complaint, and the pulmonary nodules were depicted on routine imaging examinations. However, some patients present with dyspnea and (more rarely) cyanosis, cough, pleuritic
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152
Fig. 1 a Chest CT image obtained in the pulmonary window setting showing elongated opacities with beaded appearance (arrows) in the right lower lobe of the lung. b Note also the presence of multiple small nodules with the tree-in-bud pattern (arrows)
chest pain, hemoptysis, and/or hematic sputum. Rarely, as tumor cells occupy the pulmonary arteries and arterioles, subacute cor pulmonale may manifest as progressive dyspnea, which can lead to death [8]. Exceptionally, large tumoral emboli spread from a primary tumoral mass and obstruct main pulmonary arterial vessels, creating a clinical picture indistinguishable from massive pulmonary thromboembolism [4]. Thus, the possibility of tumor embolism should be considered when a patient with known malignancy develops dyspnea. The presentation of tumor embolism on chest CT ranges from the tree-in-bud pattern to the presence of an extensive thrombus occupying the right atrium and pulmonary artery trunk associated with areas of pulmonary infarction [8]. The tree-in-bud pattern, created by centrilobular branching structures, reflects a spectrum of endo- and peribronchiolar disorders, including mucoid impaction, inflammation, and fibrosis [9]. Originally reported in cases of endobronchial spread of Mycobacterium tuberculosis, this pattern is now recognized as a CT manifestation of many diverse entities,
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Lung (2015) 193:151–153
Fig. 2 a Angio-CT of the pulmonary arteries showed that the elongated opacities represented an extensive thrombus occupying the pulmonary vessel lumen. b Thrombi were also detected in the left lower lobe of the lung
including tumoral embolism [10]. As our patient had a history of treated tuberculosis, the finding of the tree-inbud pattern raised doubt about the possibility of new infection. The finding of elongated structures with softtissue density and beaded contours proximal to the centrilobular nodules raised the suspicion of a vascular origin of this pattern. CT angiography showed that those elongated structures corresponded to thrombosed vessels proximal to the tree-in-bud pattern. Histopathologic analysis confirmed the tumoral origin of the thrombus. Although pulmonary intravascular tumor embolism is not a common entity, this possibility should be suspected in patients with known malignancy presenting with the treein-bud pattern and angiotomographic positivity for pulmonary embolism. Cor pulmonale and, less often, massive tumor embolism with acute cardiorespiratory failure may occur in these cases. Recognition of the occurrence of tumor embolism is crucial in determining the most appropriate therapeutic approach for each patient.
Lung (2015) 193:151–153
153 Conflict of interest
The authors have no conflict of interest.
References 1. Kvale PA (1991) The cancer patient with dyspnea: unusual cause? (editorial). Mayo Clin Proc 66:215–218 2. Williams & Wilkins (1990). Stedman’s Medical Dictionary. 25th edn. Williams & Wilkins, Baltimore, p. 501 3. Jakel J, Ramaswamy A, Kohler U, Barth PJ (2006) Massive pulmonary tumor microembolism from a hepatocellular carcinoma. Pathol-Res Pract 202(5):395–399 4. Martos Pe´rez F, Seara Valero R, Weil Lara B, Contreras J, Domı´nguez Pico´n F (1997) Massive tumorous pulmonary embolism in hepatocarcinoma. An Med Interna 14(12):627–629 5. Leung DY, Seah PW, Lee LC, Cranney GB, Walsh WF (1993) Embolic chondrosarcoma: an unusual cause of pulmonary embolism. Am Heart J 126(3 Pt 1):732–734 6. Alirhayim Z (2014) Chondrosarcoma Presenting as a Fatal Pulmonary Embolism (abstract). J Hosp Med 9(2):309 7. Chandrasekharan R, Bhagavaldas MC, Mathew AJ (2011) Chondrosarcoma presenting as dyspnea in a 19-year-old man: a case report. J Med Case Rep. 5:150. doi:10.1186/1752-1947-5150 8. Soares FA (1991) Morphological and clinical aspects of the vascular involvement of the pulmonary vessels by tumor emboli. J de Pneumol 17(3):127–140 9. Hansell DM, Bankier AA, MacMahon H, McLoud TC, Mu¨ller NL, Remy J (2008) Fleischner Society: glossary of terms for thoracic imaging. Radiology 246:697–722 10. Rossi SE, Franquet T, Volpacchio M, Gime´nez A, Aguilar G (2005) Tree-in-bud pattern at thin-section CT of the lungs: radiologic-pathologic overview. RadioGraphics 25:789–801
Fig. 3 a Photomicrograph showing occupation of the vascular lumen by an intravascular node composed of neoplastic cells (original magnification 1009; hematoxylin and eosin stain). b Another intravascular node comprising neoplastic cells with chondroid differentiation, characterized by stellate nuclei and abundant hyaline matrix. The remainder of the parenchyma demonstrates congestion, necrosis, and hemorrhage (original magnification 1009; hematoxylin and eosin stain)
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