LOCAL RECURRENCE OF AN ONCOCYTIC ADRENOCORTICAL CARCINOMA WITH OVARY METASTASIS
RALF KUREK, ROLF VON KNOBLOCH, ULRICH FEEK, AXEL HEIDENREICH AND RAINER HOFMANN
From the Departments of Urology and Pathology, Philipps-University, Marburg, Germany KEY WORDS: carcinoma, adrenal cortical; adenoma, oxyphilic; recurrence; adrenal glands
Oncocytic tumors of the adrenal gland are rare. To date only 18 oncocytic adrenocortical adenomas, 2 oncocytic adre- nocortical tumors of unknown malignant potential1 and 2 oncocytic adrenocortical carcinomas have been reported in the literature.2,3 We report a case of an oncocytic carcinoma that provided immunohistochemical and microsatellite data.
CASE REPORT
A 74-year-old woman presented with a large retroperito- neal mass incidentally detected by abdominal computerized tomography (CT) (fig. 1). Clinical examination and blood and urine evaluation were unremarkable. Surgery for an onco- cytic adrenocortical tumor of uncertain malignant potential on the left side had been performed 7 years earlier and the patient had received percutaneous radiation (44.2 Gy). On December 27, 1999 she underwent thoracoabdominal ne- phrectomy, splenectomy, hemicolectomy, partial pancreas re- section and right oophorectomy due to extensive infiltration of the tumor. Convalescence was uneventful.
Histological examination revealed a predominantly (approx- imately 90%) oncocytic carcinoma with isolated spongiocytic foci infiltrating the retroperitoneal fat tissue, kidney, spleen, colon and right ovary (fig. 2). Tumor cells stained positive for vimen- tin and negative for pancytokeratin monocyte nuclear factor 116 and cytokeratin 18, S100, epithelial membrane antigen, synaptophysin and chromogranin, exhibiting identical his- topathological features compared to the primary tumor re- moved 7 years earlier. Microsatellite analysis yielded loss of heterozygosity of chromosomes 3p, 9p, 13q and 17p (see table).
DISCUSSION
To our knowledge we report the first case of a local recur- rence and ovary metastasis of an oncocytic adrenocortical carcinoma. Using abdominal CT a precise differential diag- nosis of large retroperitoneal masses is sometimes difficult to Accepted for publication April 12, 2001.
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| Immunohistochemistry | Microsatellite Analysis | ||
|---|---|---|---|
| Marker | Result | Allele | Result |
| Vimentin | Pos. | 3p | Loss of heterozygosity |
| S100 | Neg. | 5q | Retained |
| Cytokeratin 18 | Neg. | 7p | Retained |
| Epithelial membrane antigen | Neg. | 9p | Loss of heterozygosity |
| Chromogranin A | Neg. | 13q | Loss of heterozygosity |
| Myocyte nuclear factor 116 | Neg. | 17p | Loss of heterozygosity |
| Synaptophysin | Neg. | 17q | Allelic imbalance |
establish. Histopathological examination of the primary tu- mor resected 7 years earlier could not specify its malignant potential and indicated only its oncocytic pattern. Only the further course of the disease led to the final diagnosis of an adrenocortical oncocytic carcinoma.
Common to all oncocytic carcinomas reported to date is neg- ativity for epithelial membrane antigen, chromogranin and S100 and positive staining for vimentin. However, immunohis- tochemical findings are heterogeneous and of little use in dif- ferential diagnosis and establishing malignancy. Microsatellite analysis revealed loss of heterozygosity at chromosome 3p as well as 9p, 13q and 17p, thereby resembling alteration patterns seen in conventional renal cell cancer and therefore of little clinical use. Thus, we recommend consideration of all oncocytic adrenocortical tumors of uncertain malignant potential to be carcinomas and performance of complete surgical resection ac- companied by a thorough clinical followup.
REFERENCES
1. Lin, B. T., Bonsib, S. M., Mierau, G. W. et al: Oncocytic adreno- cortical neoplasms: a report of seven cases and review of the literature. Am J Surg Pathol, 22: 603, 1998
2. El-Naggar, A. K., Evans, D. B. and Mackay, B .: Oncocytic adre- nal cortical carcinoma. Ultrastruct Pathol, 15: 549, 1991
3. Krishnamurthy, S., Ordonez, N. G., Shelton, T. O. et al: Fine- needle aspiration cytology of a case of oncocytic adrenocortical carcinoma. Diagn Cytopathol, 22: 299, 2000