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

Fertil Steril. 2003 Dec;80(6):1504-7. doi: 10.1016/j.fertnstert.2003.05.014.

Abstract

Objective: To report an exceptional association between an occult ovarian steroid cell tumor and a cortisol-secreting adrenal adenoma.

Design: Case report.

Setting: Endocrinology and nutrition unit at a general hospital in Spain.

Patient(s): A 49-year-old woman who presented with persistence of severe hyperandrogenism after removal of a left adrenal adenoma.

Intervention(s): 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.

Main outcome measure(s): Clinical and endocrine description of the patient and preoperative localization of the source of T secretion.

Result(s): 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.

Conclusion(s): 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.

Publication types

  • Case Reports

MeSH terms

  • Adenoma / diagnosis*
  • Adenoma / metabolism
  • Adenoma / surgery
  • Adrenal Gland Neoplasms / diagnosis*
  • Adrenal Gland Neoplasms / metabolism
  • Adrenal Gland Neoplasms / surgery
  • Adrenalectomy
  • Dehydroepiandrosterone Sulfate / blood
  • Female
  • Hirsutism / etiology*
  • Humans
  • Hydrocortisone / metabolism*
  • Hyperandrogenism / diagnosis*
  • Magnetic Resonance Imaging
  • Middle Aged
  • Neoplasms, Second Primary / diagnosis*
  • Ovarian Neoplasms / diagnosis*
  • Treatment Outcome

Substances

  • Dehydroepiandrosterone Sulfate
  • Hydrocortisone