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Sperm Retrieval and Concomitant Tumor Resection in Azoospermic Men with Congenital Adrenal Hyperplasia and Bilateral Testicular Adrenal Rest Tumors: a Case Report

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Publisher Springer
Date 2016 Feb 11
PMID 26861965
Citations 4
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Abstract

Purpose: The objective of this study was to offer a new treatment approach for sperm retrieval simultaneously with tumor resection in azoospermic men with congenital adrenal hyperplasia (CAH), orchialgia, and bilateral testicular adrenal rest tumors (TARTs) who fail to respond to medical treatment.

Methods: This is a retrospective chart review from a couple's fertility center.

Results: Between May 2013 and May 2015, two azoospermic men with CAH and bilateral TARTs, with orchialgia, and desire to conceive underwent bilateral TART resection in the same surgical setting as sperm retrieval after remaining azoospermic with normalization of gonadotropins with treatment with human chorionic gonadotropin (hCG). Both men had adequate sperm retrieved for in vitro fertilization/intracytoplasmic sperm retrieval (IVF/ICSI) at the time of bilateral TART resections. They had complete TART resections with resolution of orchialgia. The wife of one patient had a successful pregnancy with use of retrieved sperm resulting in a live birth, and the sperm from the other man is cryopreserved for future use.

Conclusions: It is feasible to perform successful sperm retrieval simultaneously with TART resection in azoospermic men with CAH after medical treatments with persistent azoospermia, rather than subjecting these men to multiple invasive procedures.

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Two cases of reversible male infertility due to congenital adrenal hyperplasia combined with testicular adrenal rest tumor.

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References
1.
Tiryaki T, Aycan Z, Hucumenoglu S, Atayurt H . Testis sparing surgery for steroid unresponsive testicular tumors of the congenital adrenal hyperplasia. Pediatr Surg Int. 2005; 21(10):853-5. DOI: 10.1007/s00383-005-1547-x. View

2.
Nicopoullos J, Ramsay J, Cassar J . From zero to one hundred million in six months: the treatment of azoospermia in congenital adrenal hyperplasia. Arch Androl. 2003; 49(4):257-63. DOI: 10.1080/01485010390204940. View

3.
Martinez-Aguayo A, Rocha A, Rojas N, Garcia C, Parra R, Lagos M . Testicular adrenal rest tumors and Leydig and Sertoli cell function in boys with classical congenital adrenal hyperplasia. J Clin Endocrinol Metab. 2007; 92(12):4583-9. DOI: 10.1210/jc.2007-0383. View

4.
Vanzulli A, DelMaschio A, Paesano P, Braggion F, Livieri C, Angeli E . Testicular masses in association with adrenogenital syndrome: US findings. Radiology. 1992; 183(2):425-9. DOI: 10.1148/radiology.183.2.1561344. View

5.
Marchini G, Cocuzza M, Pagani R, Torricelli F, Hallak J, Srougi M . Testicular adrenal rest tumor in infertile man with congenital adrenal hyperplasia: case report and literature review. Sao Paulo Med J. 2011; 129(5):346-51. PMC: 10868949. DOI: 10.1590/s1516-31802011000500010. View