Effects of mitotane on testicular adrenal rest tumors in congenital adrenal hyperplasia due to 21-hydroxylase deficiency - a retrospective series of five patients

in European Journal of Endocrinology
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  • 1 A Bachelot, Endocrinology, Groupe Hospitalier Pitié-Sapêtrière, Paris, 75013, France
  • 2 M Lapoirie, Department of Endocrinology, Hospices Civils de Lyon, Lyon, France
  • 3 J Dulon, Department of Endocrinology and Reproductive Medicine, AP-HP, IE3M, UPMC Univ Paris 06, Paris, France
  • 4 M Leban, Laboratory of endocrine biochemistry - Pitié-Salpêtrière Hospital, Assistance Publique-Hôpitaux de Paris, Sorbonne Université, Paris, 75015, France
  • 5 R Renard Penna, Radiology, Hôpital Universitaire Pitié Salpêtrière, Paris, France
  • 6 P Touraine, Endocrinology and reproductive medicine, GH Pitie Salpetriere, Paris, 75651, France

Correspondence: Anne Bachelot, Email: anne.bachelot@psl.aphp.fr
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We conducted a retrospective study on the long-term effect of mitotane treatment on testicular adrenal rest tumors (TARTs) in 5 adult patients with classic 21-hydroxylase deficiency,. After 60 months of mitotane treatment, a decrease of adrenal steroids was observed in 4 patients. Testicular ultrasonography showed complete disappearance of TART in 2 patients, stabilization in 2 patients and a halving of TART volume in the remaining patient. Sperm count improved notably in 2 patients who had normal baseline inhibin B levels and small inclusions, thus enabling cryopreservation of the subjects’ semen. Four years of follow-up of these two patients after the withdrawal of mitotane showed no recurrence of TART and persistent normal testicular function. In conclusion, mitotane should be used as a last resort in CAH patients in the cases of azoospermia associated with TARTs but normal inhibin B levels, as it can improve long-term endocrine and exocrine testicular function.


     European Society of Endocrinology

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