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Journal of Clinical Oncology, Vol 23, No 7 (March 1), 2005: pp. 1578-1579 © 2005 American Society of Clinical Oncology. DOI: 10.1200/JCO.2005.03.115
Dramatic Responses to Therapy in Rare TumorsCASE 1. Complete Remission to Corticosteroids in an Octreotide-Refractory ThymomaNational Institute for Cancer Research, Genova, Italy
San Martino Hospital, Genova, Italy
University Hospital, Parma, Italy A 64-year-old white woman with a history of recurrent oral mucositis and diarrhea associated with hypogammaglobulinemia and immunoglobulin A deficiency presented with dyspnea. Chest radiography and computed tomography (CT) showed a large mass in the upper anterior mediastinum. A radical resection was performed and histologic examination led to a diagnosis of Masaoka clinical staging system stage III epithelial thymoma (WHO classification, type B3 thymoma; prevalence of the epithelial component hematoxylin and eosin x400; Fig 1). Subsequently, the patient had consecutive palliative debulking resections for multiple local relapses. Chemotherapy (cyclophosphamide, doxorubicin, and cisplatin) was halted because of side effects (severe mucosal and gastrointestinal toxicity), probably related to the associated immunodeficiency. The patient was then referred to the Division of Medical Oncology when a CT scan of the chest and abdomen revealed progressive thoracic and hepatic metastases. Because of the high risk of toxicity from chemotherapy, and considering the intense 111In-DTPA-D-Phe1-octreotide tumor uptake, octreotide therapy was considered.1-2 Treatment with the long-acting octreotide was begun (10 mg IM every 28 days). After 6 months of treatment, a CT scan (Fig 2) demonstrated further disease progression. In the absence of alternative treatment, oral prednisone (50 mg/d) was added to octreotide therapy. Figure 3 demonstrates a dramatic complete response after 7 months of treatment with prednisone plus octreotide.
The first case of complete response to a combination of octreotide and prednisone in a patient with thymoma and pure red-cell aplasia was reported in 1997.3 However, in this case both drugs were given together from the beginning, precluding the possibility of separating the relative contribution of the two single agents in achieving tumor regression. Our case supports the important role of prednisone in determining efficacy of the octreotide-corticosteroid combination against malignant thymoma, because a complete remission was achieved only after the addition of prednisone, despite tumor progression while on prolonged octreotide treatment. However, it remains unexplained whether the impressive result is attributable to the corticosteroid itself, or the combination of octreotide and prednisone taking advantage of a possible synergistic action of the two agents, or of the ability of steroids to resume octreotide sensitivity. In the literature, single case reports of response to corticosteroids alone have been reported,4-6 primarily in cases of lymphocytic thymoma, and have been attributed to the apoptotic effects of steroids on the lymphocytic component of the tumor. However, the lymphocytic component in our case was too modest to support this simple explanation (Fig 1). Authors' Disclosures of Potential Conflicts of Interest The authors indicated no potential conflicts of interest.
REFERENCES
1. Loehrer PJ Sr, Wang W, Johnson DH, et al: Octreotide alone or with prednisone in patients with advanced thymoma and thymic carcinoma: An Eastern Cooperative Oncology Group phase II trial. J Clin Oncol 22:293-299, 2004 2. Palmieri G, Montella L, Martignetti A, et al: Somatostatin analogs and prednisone in advanced refractory thymic tumors. Cancer 94:1414-1420, 2002[CrossRef][Medline]
3. Palmieri G, Lastoria S, Colao A, et al: Successful treatment of a patient with a thymoma and pure red-cell aplasia with octreotide and prednisone. N Engl J Med 336:263-265, 1997
4. Green JD, Forman WH: Response of thymoma to steroids. Chest 65:114-116, 1974 5. Kirkove C, Berghmans J, Noel H, et al: Dramatic response of recurrent invasive thymoma to high doses of corticosteroids. Clin Oncol (R Coll Radiol) 4:64-66, 1992 6. Termeer A, Visser FJ, Mravunac M: Regression of invasive thymoma following corticosteroid therapy. Neth J Med 58:181-184, 2001[CrossRef][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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