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Journal of Clinical Oncology, Vol 23, No 16 (June 1), 2005: pp. 3844-3846 © 2005 American Society of Clinical Oncology. DOI: 10.1200/JCO.2005.05.056
Conditions Suggesting LymphomaCASE 2. Mediastinal Liposarcoma in a Patient With Previous Testicular CancerMedical Oncology Department,"S. Giovanni di Dio" Hospital, Frattamaggiore, and Experimental Pharmacology, INT Fondazione Pascale, Naples, Italy
Medical Oncology Department, "S. Giovanni di Dio" Hospital, Frattamaggiore, Naples, Italy
Department of Biochemistry and Biophysics, Second University of Naples, Naples, Italy
Thoracic Surgery Clinical Institute "Humanitas," Milan, Italy A 40-year-old man was treated in January 2002 with three cycles of adjuvant chemotherapy (BEP: bleomycin, etoposide, platinum) because of a nonseminomatous testicular cancer pathologically staged as pT1. His brother had died at age 34 years with a diagnosis of metastatic testicular seminoma. Familial and personal history was otherwise unremarkable. Patient was followed, and a chest computed tomography (CT) scan in March 2004 showed a solid and homogeneous anterior mediastinal mass measuring 7 cm at longest diameter (Fig 1, arrow). He had no symptoms. Recurrent tumor, thymic rebound, and thymic tumor were suspected. To differentiate thymic hyperplasia—frequently found in adult patients treated with chemotherapy 1,2—from other mediastinal masses, the patient underwent [111 In-DTPA-D-Phe1]-octreotide scintigraphy according to previously described methods.3 The images collected after 24 hours from tracer injection revealed uptake in the region corresponding to the left hilum, thus suggesting more a pathologic process than a benign thymic hyperplasia (Fig 2, arrow). CT-guided, fine-needle biopsy was not conclusive. Therefore, surgical intervention was carried out. At exploration, a bumpy mass measuring approximately 9 cm appeared to be closely related to left thymic horn and phrenic nerve. The mass was partly solid with cystic areas filled by mucoid material. Microscopically, atypical spindled cells similar to lipoblasts were scattered within a diffuse myxoid matrix (Figs 3A and 3B). By immunohistochemistry, vimentin and S-100 (Fig 3C) protein were expressed while carcinoembyonic antigen (Fig 3D), epithelial membrane antigen, and high and low molecular weight cytokeratins were not. The histologic appearance and immunohistochemical profile were indicative of myxoid liposarcoma. Thymic residual tissue removed with the mass was free from tumor.
Liposarcoma is the most commonly diagnosed soft tissue sarcoma in adults and occurs predominantly in the lower limbs and retroperitoneum.4 Whereas neurogenic tumors, thymoma, and lymphoma are found in approximately 65% of mediastinal tumors, mesenchymal neoplasms account for approximately 6%.4 Malignant liposarcoma develops more commonly in the posterior mediastinum. Myxoid liposarcoma accounts for 40% to 50% of all liposarcomas and has been characterized by a t(12:16)(q13-14; p11) translocation.4 Mediastinal liposarcomas are rare: there are less than 100 cases published worldwide. Liposarcomas of the anterior mediastinum to our knowledge have not been reported.5 While in early stages they are often asymptomatic, when growing to large size they exhibit various clinical symptoms mimicking lung or heart disease by compression of adjacent intrathoracic organs. This behavior may be attributed to their expansive rather than infiltrative growth. The myxoid liposarcomas seem to have a somewhat more aggressive course than the well-differentiated tumors.6 Some features displayed by CT imaging may be helpful in differentiating the fat-containing masses arising in the mediastinum.7 However, the definitive confirmation comes only from pathologic examination. In our case, somatostatin-receptor scintigraphy revealed tracer uptake by the mass. In vivo expression of somatostatin receptor was identified in 71% of 17 studied patients with metastatic sarcoma.8 However, no liposarcoma was included in this series. In vitro, one group did not find somatostatin-receptor expression in four liposarcomas by autoradiography. The different subtypes of somatostatin receptors appear to be variably expressed in a study on 31 human soft tissue tumors evaluated by reverse transcription polymerase chain reaction. In particular, four of seven liposarcomas expressed the somatostatin receptor subtype 2 mainly responsible for in vivo visualization, and two of these cases were positive for all five subtypes of somatostatin receptor. Besides the role in detection for thymic mass, [111 In-DPTA-D-Phe1]-octreotide scintigraphy could functionally define mediastinal masses of different histotypes, and its role merits further studies. The metachronous association of testicular cancer and mediastinal liposarcoma in the same patient has not been described previously. The absence of significant environmental and therapeutic risk factors (no radiotherapy and only three BEP cycles) and the concomitant occurrence of germ cell tumors in two brothers outline, once again, the need for detailed investigation in theintriguing field of cancer epidemiology, genetic diseases, and tumor antecedents. Authors' Disclosures of Potential Conflicts of Interest The authors indicated no potential conflicts of interest.
REFERENCES 1. Carmosino L, Di Benedetto A, Feffer S: Thymic hyperplasia following successful chemotherapy: A report of two cases and review of the literature. Cancer 56:1526-1528, 1985[CrossRef][Medline]
2. Mackall CL, Fleisher TA, Brown MR, et al: Age, thymopoiesis, and CD4+ T-lymphocyte regeneration after intensive chemotherapy. N Engl J Med 332:143-149, 1995
3. Lastoria S, Vergara E, Palmieri G, et al: In vivo detection of malignant thymic masses by [111 In-DTPA-D-Phe1]-octreotide scintigraphy. J Nucl Med 39:634-639, 1998 4. De Vita VT Jr, Hellman S, Rosenberg SA: Cancer: Principles and Practice of Oncology (ed 6). Philadelphia, PA, Lippincott Williams and Wilkins, 2001 5. Teschner M, Lullig H: Diagnosis and treatment of primary mediastinal liposarcoma. Pneumologie 57:22-26, 2003[Medline] 6. Klimstra DS, Moran CA, Perino G, et al: Liposarcoma of the anterior mediastinum and thymus. A clinicopathologic study of 28 cases. Am J Surg Pathol 19:782-791, 1995[Medline] 7. Gaerte SC, Meyer CA, Winer-Muram HT, et al: Fat-containing lesions of the chest. Radiographics 22: S61-S78, 2002 8. Friedberg JW, Van den Abbeele AD, Kehoe K, et al: Uptake of radiolabeled somatostatin analog is detectable in patients with metastatic foci of sarcoma. Cancer 86:1621-1627, 1999[Medline]
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Copyright © 2005 by the American Society of Clinical Oncology, Online ISSN: 1527-7755. Print ISSN: 0732-183X
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