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Anne Bachelot, Marion Lapoirie, Jerome Dulon, Monique Leban, Raphaele Renard Penna, and Philippe Touraine

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.