Mitotane treatment in patients with metastatic testicular Leydig cell tumor associated with severe androgen excess
Author(s)
Type
Journal Article
Abstract
Mitotane (o,p′DDD) is established in the adjuvant and advanced-stage treatment of adrenocortical carcinoma and counteracts both tumor growth and tumor-related steroid production. Both the adrenal glands and the gonads are steroidogenically active organs and share a common embryogenic origin. Here, we describe the effects of mitotane in two patients with metastatic Leydig cell tumor (LCT) of the testes and associated severe androgen excess (serum testosterone 93 and 88 nmol/L, respectively; male reference range 7–27 nmol/L). Both men suffered from severe restlessness, insomnia and irritability, which they described as intolerable and disrupting normal life activities. Urinary steroid profiling by gas chromatography–mass spectrometry (GC–MS) confirmed excess androgen production and revealed concurrent overproduction of glucocorticoids and glucocorticoid precursors, which under physiological conditions are produced only by the adrenal glands but not by the gonads. In a palliative approach, they were commenced on mitotane, which achieved swift control of the hormone excess and the debilitating clinical symptoms, restoring normal quality of life. GC–MS demonstrated normalization of steroid production and decreased 5α-reductase activity, resulting in decreased androgen activation, and imaging demonstrated disease stabilization for 4–10 months. In conclusion, mitotane can be highly effective in controlling steroid excess in metastatic LCTs, with anti-tumor activity in some cases.
Date Issued
2018-03-01
Date Acceptance
2018-01-08
Citation
European Journal of Endocrinology (EJE), 2018, 178 (3), pp.K21-K27
ISSN
0804-4643
Publisher
Oxford University Press
Start Page
K21
End Page
K27
Journal / Book Title
European Journal of Endocrinology (EJE)
Volume
178
Issue
3
Copyright Statement
© 2017 The authors Published by Bioscientifica Ltd. This work is licensed under a Creative Commons Attribution 4.0 International License.
License URL
Identifier
https://www.ncbi.nlm.nih.gov/pubmed/29330226
PII: EJE-17-0542
Subjects
ADRENAL-TUMORS
ADRENOCORTICAL CARCINOMA
CANCER
Endocrinology & Metabolism
EXPERIENCE
FEATURES
Life Sciences & Biomedicine
MANAGEMENT
OF-THE-LITERATURE
Science & Technology
TESTIS
THERAPY
Publication Status
Published
Coverage Spatial
England
Date Publish Online
2018-03-01