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J Thorac Cardiovasc Surg 2007;134:1371-1372
© 2007 The American Association for Thoracic Surgery

Brief Communication

Dissection of a mediastinal mature teratoma requires replacement of the ascending aorta during cardiopulmonary bypass

Satoru Kobayashi, MD, PhD*, Noriyoshi Sawabata, MD, PhD, Osamu Araki, MD, Yoko Karube, MD, PhD, Norio Seki, MD, Motohiko Tamura, MD, PhD, Hideo Umezu, MD, Hiromi Ishihama, MD, Sensuke Nagai, MD, PhD, Shinichiro Miyoshi, MD, PhD

Department of Cardiothoracic Surgery, Dokkyo University School of Medicine, Tochigi, Japan.

Received for publication March 13, 2007; accepted for publication April 23, 2007.

* Address for reprints: Satoru Kobayashi, MD, Department of Cardiothoracic Surgery, Dokkyo University School of Medicine, 880 Kitakobayashi, Mibu, Tochigi, 321-0293, Japan. (Email: kobayasi{at}dokkyomed.ac.jp).

A mediastinal mature teratoma occasionally ruptures into adjacent organs and causes various complications, although such a rupture into the ascending aorta is extremely rare. We report a case complicated by perforation of the aorta during an operation that required replacement of the aorta with an artificial graft and discuss the pathophysiology of the perforation.

Clinical Summary

A 25-year-old woman with substernal chest pain visited a local clinic in June 2003, during which time chest radiographic and electrocardiographic findings did not reveal any abnormalities. At 1 year and 3 months later, the patient visited another clinic with more severe and progressing symptoms, from which she was referred to our hospital for further investigation of an abnormal shadow on a chest X-ray image.

The patient had no notable past or family history. Systolic cardiac murmurs were auscultated in the third intercostal space in the left sternal border. A laboratory study demonstrated inflammatory findings of leukocytosis of 1.2 x 109/L, increase of C reactive protein level to 4.6 mg/dL, and increase of carcinogenetic antigen 19-9 level to 585 U/mL. Chest computed tomographic analysis revealed a cystic mass with a fat component, which was pressing the ascending aorta to the left side of the anterior mediastinum (Figure 1, A). We clinically diagnosed the lesion as a mature teratoma associated with inflammation, and a surgical resection procedure was planned.


Figure 1
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  		Figure 1. A, Computed tomographic scan revealing a fat-containing cystic mass pressing the ascending aorta to the left side. B, Operating field showing replacement of the ascending aorta with an artificial graft during cardiopulmonary bypass.

 
The operation was carried out with a median sternotomy. A cystic tumor was found originating in the thymus and was filled with dark green fluid and short hairs. Because the mass was strongly adhered to the ascending aorta through the pericardium, it was extremely difficult to peel off the portion between the tumor and the aorta. During the dissection, massive bleeding occurred because of perforation of the aorta, and because the aortic wall surrounding the perforated hole was thin and fragile, it was difficult to perform a direct suture or patch closure. As a result, the ascending aorta was replaced with an artificial graft during cardiopulmonary bypass, by using femoral artery–vein cannulation (Figure 1, B).

The resected specimen measured 9 x 7 x 4 cm in size and included a fatty mass, hairs, and fibrous tissues. Microscopic examination of the tumor revealed features of a mature teratoma with skin and adnexa, as well as fat, smooth muscle, and pancreas tissues. Necrotic tissues were also shown around the pancreas tissues (Figure 2, A).


Figure 2
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  		Figure 2. Microphotographs of the specimen. A, Pancreatic tissue (small arrow) with necrosis (large arrow) recognized in the tumor. B, Elastica–Goldner staining of the aortic wall showing severe necrosis (small arrow) that exceeded the tunica media (*) and reached to the tunica intima (#). C, Obstruction of the vasa vasorum lumens (small arrow) in the tunica adventitia.

 
Elastica–Goldner staining of the aortic wall revealed severe necrosis that exceeded the tunica media and reached to the tunica intima (Figure 2, B). Furthermore, the vasa vasorum lumens in the tunica adventitia were obstructed (Figure 2, C).

The postoperative course was uneventful, and the patient was discharged 16 days after the operation without complications.

Discussion

In a report by the Japanese Association for Thoracic Surgery in 2004, benign mediastinal germ cell tumors comprised 4.7% (149/3149) of all mediastinal tumors.1Go Benign teratomas can rupture into adjacent organs, and up to 36% of all mediastinal teratomas rupture, most frequently into the mediastinum, lung, bronchial tree, and pleural space.2Go However, life-threatening complications, such as cardiac tamponade3Go and perforation into the great vessels, including the aorta4Go or the superior vena cava, have been rarely encountered, with only a single case of perforation into the aorta known to have been reported.4Go That patient presented with exsanguinating massive hemoptysis from the aorta into the cyst and through a previously existing perforation of the cyst into a bronchus.

There are several mechanisms of tumor rupture, including autolysis by digestive enzymes released from the tumor tissues, chemical inflammation produced by sebaceous gland secretions in the tumor, ischemia caused by rapid enlargement of the tumor or increasing pressure in the cyst, and infection. In the present case a complicated perforation of the aorta occurred during the operation. The previous two occurrences of chest pain before the operation indicate that the tumor had ruptured into the mediastinum as a result of autolysis produced by digestive enzymes from the pancreatic tissues, which were identified in the tumor specimen (Figure 2, A). This inflammatory process likely led to the severe adhesion between the tumor and pericardium. Furthermore, the inflammatory reaction might have extended to the underlying ascending aorta, with subsequent weakening of the wall. Obstruction of the vasa vasorum lumens in the tunica adventitia, as shown in Figure 2, C, might also have contributed to the weakness of the aortic wall. Thus dissection of the tumor from the aorta led to perforation of the aorta. Although our patient underwent successful replacement of the perforated aorta with an artificial graft during cardiopulmonary bypass, this life-threatening complication, which can occur during surgical intervention, should be kept in mind, especially for patients with a chronic inflammatory history.

References

  1. Kazui T, Wada H, Fujita H. Thoracic cardiovascular surgery in Japan during 2003: annual report by the Japanese Association for Thoracic Surgery. Jpn J Thorac Cardiovasc Surg 2005;53:517-536.[Medline]
  2. Sasaka K, Kurihara Y, Nakajima Y, Seto Y, Endo I, Ishikawa T, et al. Spontaneous rupture: a complication of benign mature teratoma of the mediastinum. AJR Am J Roentgenol 1998;170:323-328.[Free Full Text]
  3. Marstern JL, Cooper AG, Ankeney JL. Acute cardiac tamponade due to perforation of a benign mediastinal teratoma into the pericardiac sac. Review of cardiovascular manifestations of mediastinal teratomas. J Thorac Cardiovasc Surg 1966;51:700-707.[Medline]
  4. Kolpak H. Dermoid des vorderen Mediastinum mit Perforation in die Aorta. Chirurg 1951;76:1022-1027.




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