Claahsen-van der Grinten Hedi L, Otten Barto J, Sweep Fred C G J, Hermus Ad R M M
Department of Paediatric Endocrinology, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands.
Fertil Steril. 2007 Sep;88(3):705.e5-8. doi: 10.1016/j.fertnstert.2006.11.148. Epub 2007 May 22.
To report repeated successful induction of fertility in an adult male patient with congenital adrenal hyperplasia (CAH) and testicular adrenal rest tumors (TART).
Case report.
Radboud University Nijmegen Medical Centre.
PATIENT(S): A 23-year-old male CAH patient with bilateral TART and azoospermia.
INTERVENTION(S): Changing glucocorticoid medication from 30 mg of hydrocortisone to 0.75 mg of dexamethasone (DXM) daily.
MAIN OUTCOME MEASURE(S): Improved semen analysis.
RESULT(S): 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.
CONCLUSION(S): 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.
报告一名患有先天性肾上腺增生(CAH)和睾丸肾上腺残余肿瘤(TART)的成年男性患者多次成功诱导生育的情况。
病例报告。
拉德堡德大学奈梅亨医学中心。
一名23岁的男性CAH患者,患有双侧TART且无精子症。
将糖皮质激素药物从30毫克氢化可的松改为每日0.75毫克地塞米松(DXM)。
精液分析改善。
通过超声筛查,在该23岁患者中检测到TART。精液分析显示无精子症。为了减小肿瘤大小并改善睾丸功能,将其糖皮质激素药物从氢化可的松改为等量的DXM。无精子症迅速得到逆转,其伴侣在停止口服避孕药7个月内怀孕。由于他体重逐渐增加且出现了条纹,药物又换回氢化可的松,他再次出现无精子症。两年后,患者再次开始使用相同剂量的DXM。随着体重逐渐增加他的精子浓度升高。7个月后他的伴侣第二次怀孕;之后,DXM治疗又换回氢化可的松。
在患有睾丸肾上腺残余肿瘤的男性CAH患者中,用短期等量的DXM替代氢化可的松可能会使不育症可逆。