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Background Congenital hyperplasia of the adrenal glands is a rare pathology, which can have an impact on male fertility. We report 2 cases of azoospermia in patients followed for a classical form of congenital adrenal hyperplasia. Cases presentation 1st case: After 18 months of infertility of the couple, explorations showed a high level of ACTH on the hormonal biological analysis. A therapeutic strategy combining hydrocortisone with dexamethasone induced a normal semen analysis, and the female partner of the patient subsequently had three spontaneous pregnancies. 2nd case: After two years of infertility of the couple, explorations showed adrenal testicular inclusions invading the 4/5th of the testis with a hypergonadotropic hypogonadism, the therapeutic reinforcement did not allow the improvement of semen analysis. Discussion Sertolian deficiency can be explained by: gonadotropic deficiency by excess of adrenal androgens and adrenal testicular lesions (risk of major spermatic alteration). Conclusion Congenital hyperplasia of the adrenal glands is a rare pathology in the context of male infertility. A semen analysis could be performed after puberty and a semen preservation may be proposed.

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