Abstract
We report the case of a 68-year-old woman presenting with clinical and biochemical evidence of hyperandrogenism. Imaging studies revealed the presence of a bilateral adrenal tumor. Although there was a predominant secretion of androgens, autonomous production of Cortisol was also evidenced on the basis of an inversion of the circadian rhythm of plasma Cortisol and an absence of Cortisol suppression by dexamethasone. The functional status of both tumors was demonstrated by an iodocholesterol scintigraphy and by an adrenal vein catheterization. Surgical excision of both adrenal tumors was performed and histological examination disclosed no criteria of malignancy. The patient still remains disease-free 3 years after surgery. To our knowledge, this is the second report of the bilateral occurrence of a virilizing adrenal tumor and the first in which the functional characteristics of the tumors are detailed.
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Ross N.S., Aron D.C. Hormonal evaluation of the patient with an incidentally discovered adrenal mass. N. Engl. J. Med. 323: 1401, 1990.
Harrison J.H., Mahoney E.M., Bennet A.H. Tumors of the adrenal cortex. Cancer 32: 1227, 1973.
Bertagna C., Orth D.N. Clinical and laboratory findings and results of therapy in 58 patients with adrenocortical tumors admitted to a single medical center (1951 to 1978). Am. J. Med. 71: 855, 1981.
Hintze G., Kobberling J., Becker H.D., Helmchen U., Hoffmann W. Bilateral virilizing adrenal tumor in a 17-year-old woman. J. Endocrinol. Invest. 8: 465, 1985.
Weiss L.M. Comparative histologic study of 43 metastasizing and nonmetastasizing adrenocortical tumors. Am. J. Surg. Pathol. 8: 163, 1984.
Derksen J., Nagesser S.K., Meinders A.E., Haak H.R., Van De Velde C.J.H. Identification of virilizing adrenal tumors in hirsute women. N. Engl. J. Med. 331: 968, 1994.
Luton J.P., Cerda S., Billaud L., Thomas G., Guillaume B., Bertagna X., Laudat M.H., Louvel A., Chapuis Y., Blondeau Ph., Bonnin A., Bricaire H. Clinical features of adrenocortical carcinoma, prognostic factors, and the effect of mitotane therapy. N. Engl. J. Med. 322: 1195, 1990.
Gabrilove J.L., Seman A.T., Sabet R., Mitty H.A., Nicolis G.L. Virilizing adrenal adenoma with studies of the steroid content of the adrenal venous effuent and a review of the literature. Endocr. Rev. 2: 462, 1981.
Gross M.D., Shapiro B., Freitas J.E., Ayers J., Swanson D.P., Woodbury M.C., Schteingart D.E., Beierwaltfs W.H. The relationship of 131 I-6beta-iodomethyl-19-norcholesterol (NP59) uptake to indices of androgen secretion in women with hyperandrogenism. Clin. Nucl. Med. 9: 264, 1984.
Gross M.D., Shapiro B., Francis I.R., Glazer G.M., Bree R.L., Arcomano M.A., Schteingart D.E., McLeod M.K., Sanfield J.A., Thompson N.W. Scintigraphic evaluation of clinically silent adrenal masses. J. Nucl. Med. 35: 1145, 1994.
O’Hare M.J., Monaghan P., Neville A.M. The pathology of adrenocortical neoplasia: a correlated structural and functional approach to the diagnosis of malignant disease. Hum. Pathol. 10: 137, 1979.
Hough A.J., Hollifield J.W., Page D.L., Hartmann W.H. Prognostic factors in adrenal cortical tumors. Am. J. Clin. Pathol. 72: 390, 1979.
Kamilaris T.C., De Bold C.R., Manolas K.J., Hoursanidis A., Panageas S., Yiannatos J. Testosterone-secreting adrenal adenoma in a peripubertal girl. JAMA 258: 2558, 1987.
Imai T., Seo H., Murata Y., Funahashi H., Satoh Y., Sasano H., Matsui N., Takagi H. Dexamethasone-non suppressible Cortisol in two cases with aldosterone-producing adenoma. J. Clin. Endocrinol. Metab. 72: 575, 1991.
Baert D., Nobels F., Van Crombrugge P. Combined Conn’s and Cushing’s syndrome: an unusual presentation of adrenal adenoma. Acta Clin. Belg. 50: 5, 1995.
Flecchia D., Mazza E., Carlini M., Blatto A., Olivieri F., Serra G., Camanni F., Messina M. Reduced serum levels of dehydroepiandrosterone sulphate in adrenal incidentalomas: a marker of adrenocortical tumor. Clin. Endocrinol. (Oxf.) 42: 129, 1995.
Kloos R.T., Gross M.D., Francis I.R., Korobkin M., Shapiro B. Incidentally discovered adrenal masses. Endocr. Rev. 16: 460, 1995.
Pasieka J.L., Mc Leod M.K., Thompson N.W., Gross M.D., Schteingart D.E. Adrenal scintigraphy of well-differentiated (functioning) adrenocortical carcinomas: potential surgical pitfalls. Surgery 112: 884, 1992.
Copeland P.M. The incidentally discovered adrenal mass. Ann. Intern. Med. 98: 940, 1983.
Medeiros L.J., Weiss L.M. New developments in the pathologic diagnosis of adrenal cortical neoplasms. Am. J. Clin. Pathol. 97: 73, 1992.
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Delgrange, E., Goethals, P., Laka, A. et al. An unusual case of bilateral virilizing adrenal adenoma co-secreting androgens and Cortisol. J Endocrinol Invest 19, 377–381 (1996). https://doi.org/10.1007/BF03344973
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DOI: https://doi.org/10.1007/BF03344973