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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 potential 1 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 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 Accepted for publication April 12, 2001. FIG. 1. CT demonstrates large retroperitoneal mass infiltrating left kidney, colon and spleen. FIG. 2. Adrenocortical carcinoma with predominant oncocytic dif- ferentiation. Reduced from 400. Immunohistochemical and microsatellite analyses 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 0022-5347/01/1663-0985/0 THE JOURNAL OF UROLOGY ® Vol. 166, 985, September 2001 Copyright © 2001 by AMERICAN UROLOGICAL ASSOCIATION,INC. ® Printed in U.S.A. 985

LOCAL RECURRENCE OF AN ONCOCYTIC ADRENOCORTICAL CARCINOMA WITH OVARY METASTASIS

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Page 1: LOCAL RECURRENCE OF AN ONCOCYTIC ADRENOCORTICAL CARCINOMA WITH OVARY METASTASIS

LOCAL RECURRENCE OF AN ONCOCYTIC ADRENOCORTICALCARCINOMA WITH OVARY METASTASIS

RALF KUREK, ROLF VON KNOBLOCH, ULRICH FEEK, AXEL HEIDENREICHAND 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 dateonly 18 oncocytic adrenocortical adenomas, 2 oncocytic adre-nocortical tumors of unknown malignant potential1 and 2oncocytic adrenocortical carcinomas have been reported inthe literature.2, 3 We report a case of an oncocytic carcinomathat provided immunohistochemical and microsatellite data.

CASE REPORT

A 74-year-old woman presented with a large retroperito-neal mass incidentally detected by abdominal computerizedtomography (CT) (fig. 1). Clinical examination and blood andurine evaluation were unremarkable. Surgery for an onco-cytic adrenocortical tumor of uncertain malignant potentialon the left side had been performed 7 years earlier and thepatient had received percutaneous radiation (44.2 Gy). OnDecember 27, 1999 she underwent thoracoabdominal ne-phrectomy, splenectomy, hemicolectomy, partial pancreas re-section and right oophorectomy due to extensive infiltrationof the tumor. Convalescence was uneventful.

Histological examination revealed a predominantly (approx-imately 90%) oncocytic carcinoma with isolated spongiocytic fociinfiltrating the retroperitoneal fat tissue, kidney, spleen, colonand right ovary (fig. 2). Tumor cells stained positive for vimen-tin and negative for pancytokeratin monocyte nuclear factor116 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 ofheterozygosity 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 adrenocorticalcarcinoma. Using abdominal CT a precise differential diag-nosis of large retroperitoneal masses is sometimes difficult to

establish. Histopathological examination of the primary tu-mor resected 7 years earlier could not specify its malignantpotential and indicated only its oncocytic pattern. Only thefurther course of the disease led to the final diagnosis of anadrenocortical oncocytic carcinoma.

Common to all oncocytic carcinomas reported to date is neg-ativity for epithelial membrane antigen, chromogranin andS100 and positive staining for vimentin. However, immunohis-tochemical findings are heterogeneous and of little use in dif-ferential diagnosis and establishing malignancy. Microsatelliteanalysis revealed loss of heterozygosity at chromosome 3p aswell as 9p, 13q and 17p, thereby resembling alteration patternsseen in conventional renal cell cancer and therefore of littleclinical use. Thus, we recommend consideration of all oncocyticadrenocortical tumors of uncertain malignant potential to becarcinomas 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 theliterature. 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 adrenocorticalcarcinoma. Diagn Cytopathol, 22: 299, 2000

Accepted for publication April 12, 2001.

FIG. 1. CT demonstrates large retroperitoneal mass infiltratingleft kidney, colon and spleen.

FIG. 2. Adrenocortical carcinoma with predominant oncocytic dif-ferentiation. Reduced from �400.

Immunohistochemical and microsatellite analyses

Immunohistochemistry Microsatellite Analysis

Marker Result Allele Result

Vimentin Pos. 3p Loss of heterozygosityS100 Neg. 5q RetainedCytokeratin 18 Neg. 7p RetainedEpithelial membrane antigen Neg. 9p Loss of heterozygosityChromogranin A Neg. 13q Loss of heterozygosityMyocyte nuclear factor 116 Neg. 17p Loss of heterozygositySynaptophysin Neg. 17q Allelic imbalance

0022-5347/01/1663-0985/0THE JOURNAL OF UROLOGY® Vol. 166, 985, September 2001Copyright © 2001 by AMERICAN UROLOGICAL ASSOCIATION, INC.® Printed in U.S.A.

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