Coincidental diagnosis of an occult hilar steroid cell tumor of the ovary and a cortisol-secreting adrenal adenoma in a 49-year-old woman with severe hyperandrogenism.
OBJECTIVE
To report an exceptional association between an occult ovarian steroid cell tumor and a cortisol-secreting adrenal adenoma.


METHODS
Case report.


METHODS
Endocrinology and nutrition unit at a general hospital in Spain.


METHODS
A 49-year-old woman who presented with persistence of severe hyperandrogenism after removal of a left adrenal adenoma.


METHODS
An endocrine study evaluating serum androgens, adrenal function, and tumor markers was ordered. Transvaginal sonography was done to rule out an ovarian tumor. Finally, a selective catheterization of ovarian veins allowed the correct diagnosis.


METHODS
Clinical and endocrine description of the patient and preoperative localization of the source of T secretion.


RESULTS
After adrenal surgery, urinary free cortisol levels decreased to normal values, but serum T remained within the tumoral range (3.04 ng/mL). Selective catheterization of ovarian veins revealed a gradient of T concentration in the right ovary. After bilateral annexectomy, a microscopic steroid cell tumor of hilar type was found in the right ovary. Serum T fell within the normal range, and hirsutism progressively improved.


CONCLUSIONS
This unusual association between an occult-virilizing ovarian steroid cell tumor and a cortisol-secreting adrenal adenoma illustrates the value of an accurate preoperative workup in women with severe hyperandrogenism.
Gorgojo
Juan José
JJ
Unit of Endocrinology and Nutrition, Fundación Hospital of Alcorcón, Madrid, Spain. jjgorgojo@fhalcorcon.es
Almodóvar
Francisca
F
López
Elena
E
Vicente del Cerro
Julian
J
Tejerina
Eva
E
Donnay
Sergio
S
D000236:Adenoma / Q000175:diagnosis* / Q000378:metabolism / Q000601:surgery; D000310:Adrenal Gland Neoplasms / Q000175:diagnosis* / Q000378:metabolism / Q000601:surgery; D000315:Adrenalectomy; D019314:Dehydroepiandrosterone Sulfate / Q000097:blood; D005260:Female; D006628:Hirsutism / Q000209:etiology*; D006801:Humans; D006854:Hydrocortisone / Q000378:metabolism*; D017588:Hyperandrogenism / Q000175:diagnosis*; D008279:Magnetic Resonance Imaging; D008875:Middle Aged; D016609:Neoplasms, Second Primary / Q000175:diagnosis*; D010051:Ovarian Neoplasms / Q000175:diagnosis*; D016896:Treatment Outcome
D019314:Dehydroepiandrosterone Sulfate; D006854:Hydrocortisone