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Ann Thorac Surg 1998;66:605-606
© 1998 The Society of Thoracic Surgeons

Correspondence

Recurrence of the mediastinal growing teratoma syndrome

Gilbert Massard, MDa, Françoise Eichler, MDa, Bernard Gasser, MDa, Jean-Pierre Bergerat, MDa, Jean-Marie Wihlm, MDa

a Departments of Thoracic Surgery, Medical Oncology, and Pathology, Hôpitaux Universitaires de Strasbourg, F-67091 Strasbourg, France, e-mail: Gilbert.Massard@chru-strasbourg.fr

To the Editor

In the August 1997 issue of The Annals, Dr Afifi and his colleagues from the Memorial Sloan-Kettering Cancer Center described 2 cases of the so-called mediastinal growing teratoma syndrome [1]. This name is applied to mediastinal malignant nonseminomatous germ cell tumors that show radiographic evidence of growth during or after chemotherapy, while the serum levels of tumor markers normalize during treatment. Histologic examination of the corresponding resection specimens should reveal mature teratoma without malignant components. Both reported patients had mediastinal germ cell tumors (one primary mediastinal tumor and one bulky mediastinal metastasis of a testicular tumor) with elevated {alpha}-fetoprotein and ß-human chorionic gonadotrophin levels, which rapidly normalized in response to chemotherapy. In spite of the biologic response, the tumor size progressively and paradoxically increased on radiograms. Subsequent surgical resection disclosed mature teratoma without any residual malignant remnants in both patients. To date, available publications relating such paradoxical behavior of germ cell tumors primarily concerned pulmonary metastases and some retroperitoneal tumors. Dr Afifi and his colleagues proceeded with a review of the literature, demonstrating that this phenomenon has rarely been reported together with primary mediastinal nonseminomatous germ cell tumors. We would therefore like to add the record of a patient recently operated on by our group.

A 35-year-old male patient presented in September 1995 with a large anterior mediastinal mass, which had been discovered because of a recent onset of exertional dyspnea. Computed tomographic scan showed a tumor with a transverse diameter of 19 cm. Blood samples confirmed the diagnosis of a nonseminomatous germ cell tumor: the {alpha}-fetoprotein level was 20,092 IU/mL, and the ß-human chorionic gonadotrophin level was 390 IU/mL. However, needle biopsy disclosed exclusively mature teratoma. We concluded that the mass combined both benign and malignant tissues. Chemotherapy with bleomycin, etoposide, and cisplatinum was initiated. After three cycles of treatment, the serum tumor markers had normalized. However, computed tomographic scan showed an increased tumoral mass. During the fourth cycle of chemotherapy, the patient’s dyspnea worsened because of further progression of his tumor, and hyperthermia rapidly developed. Computed tomographic scan showed marked compression of the superior mediastinum and of the large airways (Fig 1).



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  		Fig 1. Computed tomographic scan during the fourth cycle of chemotherapy (bleomycin, etoposide, cisplatinum) demonstrates an increased anterior mediastinal tumor with marked compression of the central airways; urgent resection was decided upon after this study. Histologic examination disclosed exclusively mature teratoma.

 
Progressive deterioration of the patient’s condition mandated an urgent operation on November 21, 1995. The lesion was approached through a clamshell thoracotomy and a putative complete resection was performed; a single metastasis located in the right lower lobe was resected by wedge excision. On pathologic examination, the main specimen weighed 2,300 g and measured 23 x 17 x 8 cm. All resected tissue was identified as mature teratoma. Notably, the specimen contained a marked diffuse edema, which might in part explain the apparent growth of the tumor during treatment. Postoperative recovery was delayed because of ongoing pneumonia; the patient was eventually discharged on postoperative day 22. Chemotherapy was completed postoperatively with two cycles.

A posttreatment computed tomographic scan performed in April 1996 showed a small opacity of tissular density in the vicinity of the ascending aorta, which was initially interpreted as postoperative fibrosis. This opacity was reassessed in November 1996 and proved to have increased to a transverse diameter of 35 mm; however, serum tumor marker levels remained within the normal range (Fig 2). In August 1997, the opacity had progressed to a transverse diameter of 55 mm; serum tumor markers were still within the range of normal values. Surgical reexploration was decided on, and the patient was reoperated on through a left posterolateral thoracotomy on September 17, 1997. Gross complete resection of the residual tumor was performed, with an uneventful recovery. Pathologic examination repeatedly disclosed mature teratoma without any malignancy. Complete remission was ascertained with a computed tomographic scan on January 17, 1998.



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  		Fig 2. Postoperative follow-up revealed a progressing left paraaortic mass, while serum tumor marker levels remained within the normal range. Reoperation allowed for a complete resection; histologic examination confirmed a relapsing mature teratoma.

 
An unusual feature of this additional case of growing mediastinal teratoma syndrome is the relapse of growing mature teratoma, which is most likely explained by an incomplete resection during the first operation. We are indebted to Dr Afifi and his colleagues for the definition of this syndrome and bringing it to the attention of the thoracic surgical community.

References

  1. Afifi H.Y., Bosl G.J., Burt M.E. Mediastinal growing teratoma syndrome. Ann Thorac Surg 1997;64:359-362.[Abstract/Free Full Text]



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