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Ann Thorac Surg 2007;83:1526-1528
© 2007 The Society of Thoracic Surgeons

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Case Reports

Shock Induced by Spontaneous Rupture of a Giant Thymoma

Department of Thoracic Surgery, University of Perugia, Medical School, Terni, Italy

Accepted for publication October 4, 2006.

^_* Address correspondence to Dr Puma, Chirurgia Toracica, Ospedale Civile S. Maria, Terni, 05100 Italy (Email: francescopuma{at}aospterni.it).

	Abstract

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Spontaneous bleeding of thymoma is a very rare event. We report the case of a 73-year-old woman who was referred to our hospital for acute onset of chest pain followed by shock. Chest computed tomographic scanning showed a huge mediastinal tumor with abundant left pleural effusion and contralateral shift of the mediastinum. Emergency surgical treatment was carried out through a clamshell incision. At the opening of the left pleura 1,600 mL of fresh blood was found, originating from a rupture of the tumor’s capsular veins. The lesion was completely resected, en-bloc with a wide pericardial excision. The postoperative course was uneventful. The pathology report classified the lesion as thymoma AB.

	Introduction

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Thymomas may have different clinical presentations. In nonmyastenic patients (approximately 70% of cases), thymomas are generally discovered unexpectedly on chest roentgenograms; in the remaining cases nonspecific symptoms such as cough, thoracic discomfort, or pain often lead to a delay in the diagnosis [1].

Spontaneous intralesional bleeding of a thymoma, followed by rupture, hemo-mediastinum and possibly hemothorax is exceedingly rare, and even rarer is the clinical setting of massive spontaneous bleeding that requires emergency surgical treatment.

This article reports a case of spontaneous rupture of a giant thymoma, followed by shock, managed by emergency resection through a clamshell incision.

A 73-year-old woman was referred to the emergency department of our hospital for acute onset of chest pain associated with fainting, which had started a few hours before coming to the hospital. Personal history was collected by interviewing her relatives and was basically uneventful. A few hours before admission, the patient was healthy and in good physical condition for her age. Blood samples demonstrated only anemia (9.2 g/dL) and leukocytosis (21.98 x 10³/mmc). Chest computed tomographic scanning (Fig 1) showed a huge neoplasm (12.5 x 14.5 x 12 cm) occupying the mediastinum and the left hemithorax. The density of the neoplasm was within the range of solid tissue, containing multiple, coiled vascular structures during the arterial phase and diffuse, irregular enhancement during the portal phase. The lesion was in close contact with the pulmonary artery. Abundant left pleural effusion and a partially atelectatic left lung were visible, with contralateral shift of the mediastinum. Aortic shape and dimensions were normal. With the presumptive diagnosis of bleeding from a giant sarcoma, an angiography was performed, but the study was unable to demonstrate the arterial supply to the lesion.

View larger version (86K): [in this window] [in a new window]   Fig 1. Chest computed tomographic scan shows a contralateral mediastinal shift caused by a giant neoplasm with significant heart compression and left pleural effusion. The lesion shows a rich vascularization.

View larger version (131K): [in this window] [in a new window]   Fig 2. Gross pathology specimen shows hemorrhagic areas are clearly visible on the cut surface.

The pathology report classified the lesion as thymoma AB (mixed type), with focal capsular infiltration.

Twelve months after surgery the patient is in good health and is asymptomatic. Eight months after surgery she underwent a left hemicolectomy for a sigmoid cancer.

	Comment

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Hemothorax or hemomediastinum, or both, due to a spontaneously ruptured thymoma is definitely an anecdotic event. The literature describes four cases in different reports. Caplin and colleagues [1] described one case of hemothorax, consequent to the spontaneous rupture of a thymoma in a 51-year-old man managed by emergency excision. Templeton and colleagues [2] reports a case of hemothorax due to the rupture of a malignant thymoma with infiltration of the pericardium and right atrium, presenting as an aortic dissection. Other contributions have been provided by Shimokawa and colleagues [3] and Fukuse and colleagues [4].

All the previously mentioned cases share an acute clinical onset, characterized by hemodynamic instability. Preoperative diagnosis is not always straightforward, as other causes of hemothorax or hemomediastinum or both must be ruled out [2]. In the case we observed, lesion morphology suggested a bleeding pleural fibroma or sarcoma for the high-grade vascularization, the foci of necrosis and hemorrhage, and for the concomitant hemothorax; radiological signs often characteristic of thymoma (such as calcifications and cystic components) were absent. The outstanding hemodynamic burden mandated emergency surgical management. In other case reports, surgical treatment was performed in an urgent, but not emergency setting to better define preoperative diagnosis; in Shimokawa and colleagues’ [3] experience the patient underwent delayed excision after a long period of observation and medical therapy.

In our case the dramatic clinical outbreak was triggered by bleeding of the lesion and by its huge dimensions. The sudden space-occupying effect of 1,600 mL of blood added to the bulk of the neoplasm, induced shock with a mechanism similar to cardiac tamponade.

The cause of spontaneous rupture of a thymoma is unclear. Ellison and colleagues [5] divided the mechanisms of spontaneous bleeding from a mediastinum tumor into four categories: (1) rapid lesion growth, (2) transient increase of intrathoracic pressure, (3) abrupt rise in blood pressure, and (4) altered hemostasis. Huge dimensions do not appear as a necessary condition for bleeding. Fukuse and colleagues [4] report a case of spontaneous hemorrhage in a 7-mm thymoma. In our case, hemothorax certainly derived from a rupture of subcapsular veins and thinned overlying mediastinal pleura.

In planning the surgical approach, the necessity of controlling the left pulmonary hilum was a priority. For this reason, a posterolateral thoracotomy was ruled out, and even a median sternotomy seemed unsuitable due to the tumor’s size. The clamshell incision, widely used in lung transplantation procedures, enabled rapid tumor control and an easy access to both pleural cavities and mediastinum, allowing a comfortable excision of the neoplasm as a whole.

	References

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1. Caplin JL, Gullan RW, Dymon DS, Bradley SM, Hill IM, Banim SO. Hemothorax due to rupture of a benign thymoma Jpn Heart J 1985;26:123-125.[Medline]
2. Templeton PA, Vainright JR, Rodriguez A, Diaconis JN. Mediastinal tumors presenting as spontaneous hemothorax, simulating aortic dissection Chest 1988;93:828-830.[Medline]
3. Shimokawa S, Watanabe S, Sakasegawa K, Tani A. Ruptured thymoma causing mediastinal hemorrhage resected via partial sternotomy Ann Thorac Surg 2001;71:370-372.[Abstract/Free Full Text]
4. Fukuse T, Matsukura T, Nakamura A, Kosaka S, Tamada J. Mediastinal hematoma due to thymoma hemorrhage—a case report Nippon Kyobu Geka Gakkai Zasshi 1991;39:930-934.[Medline]
5. Ellison 3rd RT, Corrao WM, Fox MJ, Braman SS. Spontaneous mediastinal hemorrhage in patient on chronic hemodialysis Ann Intern Med 1981;95:704-706.[Abstract/Free Full Text]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Mark Ragusa Francesco Puma Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Santoprete, S. Articles by Puma, F. Search for Related Content PubMed PubMed Citation Articles by Santoprete, S. Articles by Puma, F. Related Collections Mediastinum