{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,13]],"date-time":"2026-05-13T15:42:09Z","timestamp":1778686929893,"version":"3.51.4"},"reference-count":28,"publisher":"Oxford University Press (OUP)","issue":"3","license":[{"start":{"date-parts":[[2018,3,1]],"date-time":"2018-03-01T00:00:00Z","timestamp":1519862400000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["eje.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2018,3]]},"abstract":"<jats:p>Mitotane (o,p\u2032DDD) 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\u2009nmol\/L, respectively; male reference range 7\u201327\u2009nmol\/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\u2013mass spectrometry (GC\u2013MS) 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\u2013MS demonstrated normalization of steroid production and decreased 5\u03b1-reductase activity, resulting in decreased androgen activation, and imaging demonstrated disease stabilization for 4\u201310 months. In conclusion, mitotane can be highly effective in controlling steroid excess in metastatic LCTs, with anti-tumor activity in some cases.<\/jats:p>","DOI":"10.1530\/eje-17-0542","type":"journal-article","created":{"date-parts":[[2018,1,13]],"date-time":"2018-01-13T01:30:28Z","timestamp":1515807028000},"page":"K21-K27","update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-1","source":"Crossref","is-referenced-by-count":5,"title":["Mitotane treatment in patients with metastatic testicular Leydig cell tumor associated with severe androgen excess"],"prefix":"10.1093","volume":"178","author":[{"given":"Vasileios","family":"Chortis","sequence":"first","affiliation":[{"name":"1Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK"},{"name":"2Centre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicholas J","family":"Johal","sequence":"additional","affiliation":[{"name":"1Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Irina","family":"Bancos","sequence":"additional","affiliation":[{"name":"1Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK"},{"name":"3Division of Endocrinology, Metabolism and Nutrition, Mayo Clinic, Rochester, Minnesota, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matthew","family":"Evans","sequence":"additional","affiliation":[{"name":"4Departments of Pathology, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kassiani","family":"Skordilis","sequence":"additional","affiliation":[{"name":"4Departments of Pathology, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peter","family":"Guest","sequence":"additional","affiliation":[{"name":"5Radiology and Cancer Centre, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael H","family":"Cullen","sequence":"additional","affiliation":[{"name":"6Cancer Centre, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Emilio","family":"Porfiri","sequence":"additional","affiliation":[{"name":"6Cancer Centre, Queen Elizabeth Hospital, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wiebke","family":"Arlt","sequence":"additional","affiliation":[{"name":"1Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK"},{"name":"2Centre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","reference":[{"key":"ref21","first-page":"177","article-title":"Leydig cell tumors of the testis. 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