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Journal of Clinical Oncology, Vol 23, No 7 (March 1), 2005: pp. 1579-1580
© 2005 American Society of Clinical Oncology.
DOI: 10.1200/JCO.2005.03.195

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DIAGNOSIS IN ONCOLOGY

Dramatic Responses to Therapy in Rare Tumors

CASE 2. Response in a Patient With Metastatic Adrenal Cortical Carcinoma With Thalidomide

Reinaldo Chacón, Gabriela Tossen, Fernando Sanchez Loria, Matías Chacón

University Hospital, Parma, Italy
Medical and Surgical Oncology Departments, Alexander Fleming Institute, Buenos Aires, Argentina

A 40-year-old female presented with abdominal pain. A computed tomography (CT) scan showed a 17 x 16 cm heterogeneous mass in the right adrenal, which was resected. She had no previous history of other neoplasia or family hereditary syndromes. Pathology showed a high-grade adrenocortical carcinoma, vimentin positive, with vascular involvement and no capsular invasion or nodal disease. Adjuvant therapy with six cycles of epirubicin and cisplatin were administered. Two years later, a CT scan showed at least three liver lesions. A biopsy was performed and metastatic adrenal carcinoma was confirmed. The patient developed progressive disease after three cycles of carboplatin and etoposide, and 4 months of mitotane (Fig 1). She was started on thalidomide 100 mg per day, without side effects during the first month. The dose was then increased to 200 mg per day. After 5 months of treatment, she had gained 10 kg, and CT scan (Fig 2) showed partial response, which was a marked improvement from the CT scan of 6 months earlier (Fig 3). As of the publication date, she is asymptomatic and continues to take thalidomide 200 mg per day.



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Fig 1.
 


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Fig 2.
 


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Fig 3.
 
Adrenal cortical carcinoma (ACC) is a rare cancer, and accounts for less than 0.2% of all cancers. The mainstay of treatment is complete surgical resection. Adjuvant treatments have not been proven to prolong disease-free survival or overall survival. In advanced disease, mitotane- or cisplatin-based chemotherapy regimens are widely used; the response rate is less than 20%, and there have been few long-term survivors with metastatic adrenal carcinoma.1,2,3

The inhibition of new blood vessel growth is an active area of research in cancer therapy.4 Today, thalidomide (historically a highly teratogenic sedative) is an active drug in Kaposi's sarcoma, myeloma, and cachexia syndrome, and is less effective in advanced renal carcinoma. One objective of such therapies is to downregulate the vascular endothelial growth factor. There is no available data on ACC treated with antiangiogenic drugs. This case shows an excellent imaging response with thalidomide in a very resistant tumor (ACC).

Future studies will be required to demonstrate if the antiangiogenic properties of thalidomide are responsible for the volume decrease in chemoresistant tumors.

Authors' Disclosures of Potential Conflicts of Interest

The authors indicated no potential conflicts of interest.

REFERENCES

1. Van Slooten H, Moolenaar AJ, Van Seters AP: The treatment of adrenocortical carcinoma with o,p'-DDD: Prognostic implications of serum levels monitoring. Eur J Cancer Clin Oncol 20:47-53, 1984[CrossRef][Medline]

2. Haak HR, Hermans J, Van de Velde CS: Optimal treatment of adrenocortical carcinoma with mitotane: Results in a consecutive series of 96 patients. Br J Cancer 69:947-951, 1994[Medline]

3. Bonacci R, Gigliotti A, Baudin E: Cytotoxic therapy with etoposide and cisplatin in advanced adrenocortical carcinoma: Reseau Comete INSERM. Br J Cancer 78:546-549, 1998[Medline]

4. D'Amato RJ, Loughnan MS, Flynn E, et al: Thalidomide is an inhibitor of angiogenesis. Proc Natl Acad Sci U S A 91:4082-4085, 1994[Abstract/Free Full Text]


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