Hyperandrogenism due to a testosterone-secreting Sertoli-Leydig cell tumor associated with a dehydroepiandrosterone sulfate-secreting adrenal adenoma in a postmenopausal woman: case presentation and review of literature

Endocr Pract. 2009 Mar;15(2):149-52. doi: 10.4158/EP.15.2.149.

Abstract

Objective: To report a case of hyperandrogenism attributable to the presence of an adrenal adenoma secreting dehydroepiandrosterone sulfate (DHEA-S) and an ovarian Sertoli-Leydig cell tumor secreting testosterone in a postmenopausal woman.

Methods: The laboratory, radiologic, and pathologic findings in our case are described. In addition, the pertinent literature is reviewed.

Results: A 56-year-old woman presented with a history of gradual increase in facial and body hair, scalp hair loss, male pattern baldness, and deepening of her voice, beginning a few years after spontaneous menopause at age 49 years. She had hypertension, obesity, and type 2 diabetes mellitus. Laboratory tests showed elevated levels of total testosterone (348 ng/dL) and DHEA-S (2,058 microg/dL), and a left adrenal tumor (3 by 4 cm) was detected on abdominal computed tomographic scan. Laparoscopic left adrenalectomy was performed, and the pathologic diagnosis was adrenal adenoma. The DHEA-S returned to normal levels, but the serum testosterone concentration remained elevated. Transvaginal ultrasonography disclosed an ovarian tumor. Bilateral oophorectomy was performed, and an ovarian Sertoli-Leydig cell tumor was diagnosed. The hormonal and clinical picture normalized after this surgical intervention.

Conclusion: After extensive review of the literature, we believe that this is the first reported case of a coincidental DHEA-S-secreting adrenal adenoma and a testosterone- secreting ovarian Leydig cell tumor causing signs of virilization.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adrenocortical Adenoma / metabolism*
  • Adrenocortical Adenoma / pathology
  • Adrenocortical Adenoma / physiopathology
  • Alopecia
  • Dehydroepiandrosterone Sulfate / metabolism*
  • Female
  • Humans
  • Hyperandrogenism / diagnosis*
  • Hyperandrogenism / etiology*
  • Hyperandrogenism / pathology
  • Leydig Cell Tumor / metabolism*
  • Leydig Cell Tumor / pathology
  • Leydig Cell Tumor / physiopathology
  • Middle Aged
  • Postmenopause
  • Testosterone / metabolism*

Substances

  • Testosterone
  • Dehydroepiandrosterone Sulfate