Testicular adrenal rest tumors in adult males with congenital adrenal hyperplasia: evaluation of pituitary-gonadal function before and after successful testis-sparing surgery in eight patients
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CONTEXT: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors (TART) are frequently present. These tumors can interfere with testicular function. Intensifying glucocorticoid therapy does not always lead to tumor regression and improvement of testicular function. Recently, testis-sparing surgery was introduced for treatment of TART. OBJECTIVE: The aim of this study was to evaluate tumor volume, symptoms, and pituitary-gonadal function in male patients with CAH caused by 21-hydroxylase deficiency and bilateral TART before and after testis-sparing surgery. SETTING: This study was conducted at Radboud University Nijmegen Medical Centre in The Netherlands. PATIENTS: Eight adult male CAH patients with bilateral TART and infertility were included. INTERVENTIONS: Evaluation of testicular magnetic resonance imaging, symptoms, fasting serum concentrations of ACTH, LH, FSH, inhibin B, 17-OH progesterone, androstenedione, testosterone, and estrone, and semen analysis (six of eight patients) was performed before and 6 and 22 months after testis-sparing surgery. MAIN OUTCOME MEASURES: The main outcome measures were absence of residual tumor and improvement of symptoms and pituitary-gonadal function. RESULTS: Residual tumors were not found on any of the patients' magnetic resonance imaging after surgery. Two patients reported testicular pain and discomfort that disappeared after surgery. Parameters of pituitary-gonadal function did not improve after surgery: semen analysis showed azoospermia (five patients) or oligospermia (one patient) without improvement, and all patients had persistently low inhibin B concentrations. CONCLUSION: Testis-sparing surgery did not improve pituitary-gonadal function despite successful removal of the tumors. Further studies are needed to investigate whether surgery at an earlier stage in the natural history of TART can prevent permanent testicular damage.
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Testis-preserving surgery with TART resection has not restored fertility.
“Testis-preserving surgery with TART resection has not restored fertility, 123”
From Congenital adrenal hyperplasia (2017) · cites this paper as [123]
The prevalence of testicular adrenal rest tumors in boys aged 2 to 18 years with classic CAH varies from 21% to 28%.
“The prevalence of TARTs in boys aged 2 to 18 years who have classic CAH varies from 21% to 28% (227, 251); there have been few studies describing TARTs in males with NCCAH.”
From Congenital Adrenal Hyperplasia Due to Steroid 21-Hydroxylase Deficiency: An Endocrine Society* Clinical Practice Guideline (2018) · cites this paper as [227]
Men with large testicular adrenal rest tumors may have low morning testosterone, indicating poor Leydig cell function.
“Men with large TARTs may also have low morning testosterone, indicating poor Leydig cell function (227).”
From Congenital Adrenal Hyperplasia Due to Steroid 21-Hydroxylase Deficiency: An Endocrine Society* Clinical Practice Guideline (2018) · cites this paper as [227]
Testicular adrenal rest tumors are usually small and bilateral, not palpable, but easily detectable by ultrasound.
“TARTs are usually small and bilateral, not palpable, but easily detectable by ultrasound (227, 251).”
From Congenital Adrenal Hyperplasia Due to Steroid 21-Hydroxylase Deficiency: An Endocrine Society* Clinical Practice Guideline (2018) · cites this paper as [227]
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- Authors
- Claahsen-van der Grinten, H. L., Otten, B. J., Takahashi, S., Meuleman, E. J. H., Hulsbergen-van de Kaa, C., Sweep, F. C. G. J., & Hermus, A. R. M. M.
- Affiliations
- Department of Paediatric Endocrinology (H.L.C.-v.d.G., B.J.O.), Radboud University Nijmegen Medical Centre, 6500 HB Nijmegen, The Netherlands
- Radboud University Medical Center (NL)
- Department of Pathology (C.H.-v.d.K.), Radboud University Nijmegen Medical Centre, 6500 HB Nijmegen, The Netherlands
- Venue
- The Journal of Clinical Endocrinology & Metabolism
- Publisher
- The Endocrine Society
- Published
- 2006-11-07
- Volume
- 92
- Issue
- 2
- Pages
- 612-615
- ISSN
- 0021-972X (ISSN-L)
- 1945-7197
- OpenAlex
- W2114063488
- Cited by
- 139
- References
- 20
- Citations by year
- 2026: 6 · 2025: 4 · 2024: 3 · 2023: 8 · 2022: 7 · 2021: 8 · 2020: 6 · 2019: 7 · 2018: 3 · 2017: 7 · 2016: 3 · 2015: 13 · 2014: 11 · 2013: 4 · 2012: 8
- FWCI
- 2.85
- Type
- journal article
- Language
- en
- Open access
- green
- hdl.handle.net
- Licence
- other-oa
- Locations
- The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
- PubMed (publishedVersion)
- Radboud Repository (Radboud University) (submittedVersion)
- Radboud Repository (Radboud University) (submittedVersion, other-oa)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Sexual Differentiation and Disorders
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Adrenal Hormones and Disorders
- Health Sciences › Medicine › Surgery › Testicular diseases and treatments
- Keywords
- testicular adrenal rest tumor, congenital adrenal hyperplasia, 21-hydroxylase deficiency, testis-sparing surgery, pituitary-gonadal function, male infertility, azoospermia, inhibin B, tumor regression, glucocorticoid therapy, FSH and LH
- MeSH terms
- Adrenal Hyperplasia, Congenital, Adrenal Rest Tumor, Adrenocorticotropic Hormone, Adult, Androstenedione, Biopsy, Estrone, Follicle Stimulating Hormone, Humans, Inhibins, Luteinizing Hormone, Magnetic Resonance Imaging, Male, Pituitary Gland, Postoperative Complications, Preoperative Care, Urologic Surgical Procedures, Male, Testicular Neoplasms, Testis, Testosterone, Treatment Outcome, 17-alpha-Hydroxyprogesterone, Azoospermia
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