Repeated successful induction of fertility after replacing hydrocortisone with dexamethasone in a patient with congenital adrenal hyperplasia and testicular adrenal rest tumors.
OBJECTIVE
To report repeated successful induction of fertility in an adult male patient with congenital adrenal hyperplasia (CAH) and testicular adrenal rest tumors (TART).


METHODS
Case report.


METHODS
Radboud University Nijmegen Medical Centre.


METHODS
A 23-year-old male CAH patient with bilateral TART and azoospermia.


METHODS
Changing glucocorticoid medication from 30 mg of hydrocortisone to 0.75 mg of dexamethasone (DXM) daily.


METHODS
Improved semen analysis.


RESULTS
With the use of ultrasound screening, TART were detected in the 23-year-old patient. The semen analysis showed azoospermia. In an attempt to decrease tumor size and improve testicular function, his glucocorticoid medication was changed from hydrocortisone to an equivalent dosage of DXM. The azoospermia was quickly reversed, and his partner conceived within 7 months of stopping oral contraceptives. Due to his progressive weight gain and striae, his medication was changed back to hydrocortisone, and he again developed again azoospermia. Two years later, the patient started again with DXM at the same dosage. His sperm concentration increased with progressive weight gain. Seven months later his partner became pregnant for the second time; afterward, the DXM treatment was changed back to hydrocortisone.


CONCLUSIONS
In male CAH patients with testicular adrenal rest tumors, infertility may be reversible by replacing hydrocortisone with short courses of an equivalent dosage of DXM.
Claahsen-van der Grinten
Hedi L
HL
Department of Paediatric Endocrinology, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands. h.claahsen@cukz.umcn.nl <h.claahsen@cukz.umcn.nl>
Otten
Barto J
BJ
Sweep
Fred C G J
FC
Hermus
Ad R M M
AR
D000312:Adrenal Hyperplasia, Congenital / Q000150:complications* / Q000188:drug therapy; D000328:Adult; D000728:Androgens / Q000032:analysis; D003907:Dexamethasone / Q000627:therapeutic use*; D005298:Fertility / Q000187:drug effects / Q000502:physiology*; D006801:Humans; D006854:Hydrocortisone / Q000627:therapeutic use*; D008297:Male; D012463:Saliva / Q000737:chemistry; D013076:Sperm Count; D013736:Testicular Neoplasms / Q000150:complications* / Q000188:drug therapy
D000728:Androgens; D003907:Dexamethasone; D006854:Hydrocortisone