Ann Thorac Surg 2008;86:1971-1973. doi:10.1016/j.athoracsur.2008.05.003
© 2008 The Society of Thoracic Surgeons
Case Reports
Cystic Malignant Teratoma With Early Recurrence After Intraoperative Spillage
Chin-Chih Chang, MDa,
Yih-Leong Chang, MDb,
Yung-Chie Lee, MD, PhDa,*
a Department of Surgery, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan
b Department of Pathology, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan
Accepted for publication May 5, 2008.
* Address correspondence to Dr Lee, Department of Surgery, National Taiwan University Hospital, No. 7, Chung-Shan South Rd, Taipei 100, Taiwan (Email: ntuhycl{at}gmail.com).
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Abstract
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Teratoma with malignant transformation, defined as germ cell tumors with a malignant component of nongerm cell tumors, such as sarcomas and carcinomas, is rarely seen in the mediastinum. A rare case of the mediastinal mature cystic teratoma with focal gastrointestinal adenocarcinoma is described in this report. The patient underwent a video-assisted thoracoscopic surgery for tumor resection. Intraoperatively, the cystic tumor was ruptured due to dense adhesion. The rapid tumor dissemination was postoperatively noted. Careful and complete resection with the cyst wall of the mediastinal mature cystic teratoma intact is necessary to avoid a risk of relapse from a potentially malignant component.
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Introduction
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Teratoma with malignant transformation (TMT) is defined as germ cell tumors with a malignant component of nongerm cell tumors (eg, sarcomas or carcinomas). TMT, mostly found in the gonadal area, is rare and occurs in approximately 2% of gonadal mature cystic teratoma [1]. Mediastinal TMT is extremely rare. Rupture of TMT can cause rapid dissemination with a poor prognosis [2, 3]. We present a rare case of gastrointestinal adenocarcinoma arising in a mediastinal mature cystic teratoma with the intraoperative rupture of the tumor, followed by rapid postoperative relapse.
A 52-year-old woman accidentally found the mediastinal mass in a medical check-up. Chest computed tomographic scan showed a lobulated and cystic mass with calcified and fat components in the right-sided paratracheal mediastinum (Fig 1A). Preoperative serum carcinoembryonic antigen was within normal limits. After a preliminary diagnosis of benign mature cystic teratoma was made, video-assisted thoracoscopic surgery (VATS) for the tumor excision was performed. Intraoperatively, the tumor was ruptured due to dense adhesion to the mediastinum, and yellowish sebaceous fluid spilled out to the pleural cavity. The pathologic study described a 6.5 x 5.3 x 3.0 cm cystic mass, mostly composed of mature ectodermal and mesodermal elements, including cartilage, columnar epithelium, and fibro-adipose tissue (Fig 2A). Only a small and solid part of the tumor was poorly differentiated adenocarcinoma. The signet-ring-shaped tumor cells were strongly positive for cytokeratin 20 (Fig 2B) and mucicarmine staining, but negative for cytokeratin 7 and thyroid transcription factor-1 protein. This result supported the diagnosis of a gastrointestinal adenocarcinoma. The postoperative course was uneventful and the patient was discharged on postoperative day 4. Unfortunately, at the 3-month follow-up, the chest roentgenogram evidenced right-sided mediastinal widening. The chest computed tomographic scan revealed a large mediastinal mass in the right-sided paratracheal region, which encased the right brachiocephalic vein. Besides, focal parietal pleural nodules were also discovered (Fig 1A). Because of the spread of the recurrent tumors, the patient underwent chemotherapy and it worked well. One year after the operation, she was alive with stable condition.
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Fig 1. (A) Preoperative chest computed tomographic (CT) scan showed a lobulated mass with cystic and calcified solid components in the right-sided paratracheal mediastinum. (B) The follow-up chest CT scan 3 months after operation demonstrated a large mediastinal mass in the right-sided paratracheal region with parietal pleural nodules and encasement of right brachiocephalic vein.
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Fig 2. (A) Histologic finding of the tumor displayed a mature teratoma composed chiefly of columnar epithelium, cartilage, and fibro-adipose tissue. (Hematoxylin and eosin; x33.) (B) A small solid area of the tumor disclosed a poorly differentiated gastrointestinal adenocarcinoma, which was strongly immunoreactive to cytokeratin 20. (Avidin Biotin Complex method; x33.)
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Comment
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Mediastinal TMT is extremely rare. The TMT is summarized in two types: (1) chemotherapy-induced or irradiation-induced TMT and (2) naturally occurring TMT. The former type occurs in young patients with an initially malignant germ cell tumor, whereas the latter in older ones with mature teratomas [4]. Naturally occurring TMTs of the mediastinum with precise histopathology were reported in a few cases [1, 3, 4]. The incidence of mediastinal TMT can be calculated according to previous large series of mediastinal teratomas in 1.7% (1 of 58) [5] to 8.0% (11 of 138) [6]. Dulmet and colleagues [5] reported only 1 patient of an immature teratoma with an embryonal rhabdomyosarcoma in 58 patients of mediastinal teratomas. Moran and colleagues [6] reported 4 cases of carcinomas and 7 of sarcomas in 138 cases of mediastinal teratomas. However, the precise clinical history and course of these patients are unclear; thus, we can not know which of the patients belonged to the type of naturally occurring TMT.
Gastrointestinal adenocarcinoma arising in mature cystic teratoma is extremely rare. The malignant component of naturally occurring TMT is exclusively carcinomatous [4]. Squamous cell carcinoma accounts for 69% to 85% of the ovarian TMT, and adenocarcinoma occurs in 7% of cases [1, 7, 8]. A variety of histologic origins of adenocarcinoma in ovarian mature cystic teratoma have been reported, including epithelia of mammary, salivary, sebaceous, thyroid glands, and epithelia of gastrointestinal and respiratory tracts [7, 8]. We believe that only three cases of adenocarcinoma arising in the mediastinal teratoma have been reported in the English literature [1, 3, 4], and one of them originates from the gastrointestinal cells [1]. Cytokeratin immunohistochemical staining can help differentiate gastrointestinal adenocarcinoma from other histologic origins of adenocarcinoma. Gastrointestinal adenocarcinoma is usually positive for cytokeratin 20 and negative for cytokeratin 7.
Complete resection with the cyst wall intact has been reported to be an important prognostic factor in TMT [1, 4, 7, 8]. We previously reported a case of a giant mature cystic teratoma with a focal gastrointestinal adenocarcinoma in the posterior mediastinum, extending into the retroperitoneum. The patient underwent complete surgical resection, and no evidence of tumor recurrence was noted for more than 2 years [1]. However, the prognosis was poor in the cases of tumor rupture [2, 3]. Jung and associates [3] reported that one case of mediastinal teratoma with poorly differentiated adenocarcinoma, complicated by spontaneous rupture, died 6 months after the surgery. Successful VATS resection of benign mediastinal cystic teratomas has been reported [9–12]. However, due to the characteristic of dense adhesion of benign teratomas to adjacent structures, conversion to an open resection is necessary when unexpected problems or difficulties are found during VATS to minimize the risk of intraoperative spillage of the cystic contents.
In conclusion, although mediastinal TMTs are extremely rare encounters, it should be suspected in all clinically diagnosed mature cystic teratomas presumed to be benign. Avoiding intraoperative spillage of mediastinal teratoma should be fully implemented to minimize the risk of recurrence of malignant components.
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References
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Abstract
Introduction
