Testicular adrenal rest tumours in young adult males with congenital adrenal hyperplasia: prevalence and impact on testicular function
Abstract
Testicular adrenal rest tumours (TARTs) have been described in patients with congenital adrenal hyperplasia (CAH). The aim of the study was to determine the prevalence of TARTs in patients with CAH, the associated factors and their impact on gonadal function. It is a prospective study concerning six young adult men with CAH, four cases with 21-hydroxylase deficiency and two cases with 11-hydroxylase deficiency. All patients were under glucocorticoid therapy. The mean age was 25 years (range: 20-31). All patients underwent a physical examination with testicular palpation, scrotal ultrasonography, a blood sample for serum testosterone, FSH, LH, inhibin B, ∆4-androstenedione and 17-OH-progesterone measurements and a semen analysis. Ultrasound revealed TARTs in four patients; three were bilateral. The mean tumour size was 6.3 ml (range: 0.02-14.1). The tumours were palpable in two cases. 17-OH-progesterone was <10 ng/ml in all cases. Decreased testosterone level was found in one case. The semen analysis revealed azoospermia in one case and poor semen quality in four patients. TARTs were common and associated with impaired spermatogenesis.
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TART has also been reported in 11βOH and HSD3B2 deficiency.
“and has also been reported in 11βOH and HSD3B2 deficiency, 117-119”
From Congenital adrenal hyperplasia (2017) · cites this paper as [119]
Cites
Congenital adrenal hyperplasia due to 21-hydroxylase deficiency*
White, P. C., & Speiser, P. W. (2000) · Endocrine Reviews · doi:10.1210/edrv.21.3.0398 · Cited by 1,432 · see citing works
Congenital Adrenal Hyperplasia due to 21-Hydroxylase Deficiency
White, P. C. (2000) · Endocrine Reviews · doi:10.1210/er.21.3.245 · Cited by 877 · see citing works
Health status of adults with congenital adrenal hyperplasia: a cohort study of 203 patients
Arlt, W., Willis, D. S., Wild, S. H., Krone, N., Doherty, E. J., Hahner, S., Han, T. S., Carroll, P. V., Conway, G. S., Rees, D. A., et al. (2010) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2010-0917 · Cited by 506 · see citing works
Stikkelbroeck, N. M. M. L., Otten, B. J., Pasic, A., Jager, G. J., Sweep, C. G. J. F., Noordam, K., & Hermus, A. R. M. M. (2001) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.86.12.8090 · Cited by 305 · see citing works
The Testicular “Tumor” of the Adrenogenital Syndrome
Rutgers, J. L., Young, R. H., & Scully, R. E. (1988) · The American Journal of Surgical Pathology · doi:10.1097/00000478-198807000-00001 · Cited by 188 · see citing works
Testicular adrenal rest tumours in congenital adrenal hyperplasia
Claahsen-van der Grinten, H. L., Otten, B. J., Stikkelbroeck, M. M. L., Sweep, F. C. G. J., & Hermus, A. R. M. M. (2009) · Best Practice & Research Clinical Endocrinology & Metabolism · doi:10.1016/j.beem.2008.09.007 · Cited by 187 · see citing works
Wilkins, L., Fleischmann, W., & Howard, J. E. (1940) · Endocrinology · doi:10.1210/endo-26-3-385 · Cited by 176 · see citing works
High prevalence of reduced fecundity in men with congenital adrenal hyperplasia
Reisch, N., Flade, L., Scherr, M., Rottenkolber, M., Pedrosa Gil, F., Bidlingmaier, M., Wolff, H., Schwarz, H.-P., Quinkler, M., Beuschlein, F., et al. (2009) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2008-1414 · Cited by 170 · cited 2× · see citing works
“Similarly, the size of a TART can differ according to the type of CAH (Reisch et al., 2009).”
BackgroundResultFalhammar, H., Nyström, H. F., Ekström, U., Granberg, S., Wedell, A., & Thorén, M. (2012) · European Journal of Endocrinology · doi:10.1530/eje-11-0828 · Cited by 170 · see citing works
der Grinten, H. L. C., Sweep, F. C. G. J., Blickman, J. G., Hermus, A. R., & Otten, B. J. (2007) · European Journal of Endocrinology · doi:10.1530/eje-07-0201 · Cited by 142 · see citing works
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. (2007) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2006-1311 · Cited by 139 · see citing works
Stikkelbroeck, N. M. M. L., Suliman, H. M., Otten, B. J., Hermus, A. R., Blickman, J. G., & Jager, G. J. (2003) · European Radiology · doi:10.1007/s00330-002-1786-3 · Cited by 126 · see citing works
der Grinten, H. L. C., Otten, B. J., Sweep, F. C. G. J., Span, P. N., Ross, H. A., MEULEMAN, E. J. H., & Hermus, A. R. (2007) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2007-0337 · Cited by 123 · see citing works
Martinez-Aguayo, A., Rocha, A., Rojas, N., García, C., Parra, R., Lagos, M., Valdivia, L., Poggi, H., & Cattani, A. (2007) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2007-0383 · Cited by 114 · see citing works
Nermoen, I., Rørvik, J., Holmedal, S. H., Hykkerud, D. L., Fougner, K. J., Svartberg, J., Husebye, E. S., & Løvås, K. (2011) · Clinical Endocrinology · doi:10.1111/j.1365-2265.2011.04151.x · Cited by 103 · cited 1× · see citing works
“Two studies suggested a positive correlation between ACTH and 17-OH-progesterone and the frequency of TARTs (Nermoen et al., 2011; Pierre et al., 2012).”
BackgroundClark, R. V., Albertson, B. D., Munabi, A. K., CASSORLA, F. G., Aguilera, G., Warren, D. W., Sherins, R. J., & Loriaux, D. L. (1990) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem-70-5-1408 · Cited by 99 · see citing works
Aycan, Z., Bas, V. N., Cetinkaya, S., Yilmaz Agladioglu, S., & Tiryaki, T. (2013) · Clinical Endocrinology · doi:10.1111/cen.12033 · Cited by 98 · see citing works
Reisch, N., Scherr, M., Flade, L., Bidlingmaier, M., Schwarz, H.-P., Müller-Lisse, U., Reincke, M., Quinkler, M., & Beuschlein, F. (2010) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2009-1929 · Cited by 92 · see citing works
Reisch, N., Rottenkolber, M., Greifenstein, A., Krone, N., Schmidt, H., Reincke, M., Schwarz, H.-P., & Beuschlein, F. (2013) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2012-3181 · Cited by 82 · see citing works
Massive Hyperplasia of Testicular Adrenal Rests in a Patient with Nelson’s Syndrome
Johnson, R. E., & Scheithauer, B. W. (1982) · American Journal of Clinical Pathology · doi:10.1093/ajcp/77.4.501 · Cited by 68 · see citing works
Cunnah, D., Perry, L., DACIE, J. A., Grant, D., Lowe, D., Savage, M. O., & BESSER, G. M. (1989) · Clinical Endocrinology · doi:10.1111/j.1365-2265.1989.tb03735.x · Cited by 62 · see citing works
Testicular Adrenal Rest Tumors in Patients With Congenital Adrenal Hyperplasia
Delfino, M., Elia, J., Imbrogno, N., Argese, N., Mazzilli, R., Toscano, V., & Mazzilli, F. (2012) · Journal of Ultrasound in Medicine · doi:10.7863/jum.2012.31.3.383 · Cited by 52 · see citing works
Testicular Hilar Nodules in Adrenogenital Syndrome
Shanklin, D. R. (1963) · American journal of diseases of children · doi:10.1001/archpedi.1963.02080050245001 · Cited by 51 · see citing works
Mouritsen, A., Jørgensen, N., Main, K. M., Schwartz, M., & Juul, A. (2009) · International Journal of Andrology · doi:10.1111/j.1365-2605.2009.00967.x · Cited by 48 · see citing works
Testicular Adrenal Rest Tumors in Patients with Congenital Adrenal Hyperplasia
Çakır, E. D. P., Mutlu, F. Ş., Eren, E., Paşa, A. Ö., Sağlam, H., & Tarım, Ö. (2012) · Journal of Clinical Research in Pediatric Endocrinology · doi:10.4274/jcrpe.563 · Cited by 35 · see citing works
Pierre, P. R., Despert, F., Tranquart, F., Coutant, R., Tardy, V., Kerlan, V., Sonnet, E., Baron, S., LORCY, Y., Émy, P., et al. (2012) · Annales d Endocrinologie · doi:10.1016/j.ando.2012.09.005 · Cited by 31 · see citing works
Kang, M. J., Kim, J. H., Lee, S. H., Lee, Y. A., Shin, C. H., & Yang, S. W. (2011) · Endocrine Journal · doi:10.1507/endocrj.k11e-034 · Cited by 25 · see citing works
Bayhan, G. İ., Çetinkaya, S. Ç., Çinar, H. G., & Aycan, Z. (2010) · Journal of Pediatric Endocrinology and Metabolism · doi:10.1515/jpem.2010.23.7.729 · Cited by 5 · see citing works
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El-Maouche, D., Arlt, W., & Merke, D. P. (2017) · The Lancet · doi:10.1016/s0140-6736(17)31431-9 · Cited by 669 · see citing works
Engels, M., Span, P. N., van Herwaarden, A. E., Sweep, F. C. G. J., Stikkelbroeck, N. M. M. L., & Claahsen-van der Grinten, H. L. (2019) · Endocrine Reviews · doi:10.1210/er.2018-00258 · Cited by 163 · see citing works
Bachelot, A., Lapoirie, M., Dulon, J., Leban, M., Renard Penna, R., & Touraine, P. (2021) · European Journal of Endocrinology · doi:10.1530/eje-20-0787 · Cited by 19 · see citing works
- Authors
- Chihaoui, M., Kanoun, F., Chaker, F., Yazidi, M., Bouchrit, K., Mizouni, H., Feki, M., Kharrat, M., & Slimane, H.
- Affiliations
- Hôpital La Rabta (TN)
- Faculté de médecine de Tunis (TN)
- Corresponding author
- Chihaoui, M.
- Venue
- Andrologia
- Publisher
- Hindawi Limited
- Published
- 2015-04-16
- Volume
- 48
- Issue
- 1
- Pages
- 45-50
- ISSN
- 0303-4569 (ISSN-L)
- OpenAlex
- W1988596920
- Cited by
- 23
- References
- 28
- Citations by year
- 2026: 1 · 2025: 1 · 2024: 3 · 2023: 4 · 2022: 2 · 2021: 4 · 2020: 1 · 2019: 2 · 2018: 2 · 2017: 2 · 2016: 1
- FWCI
- 0.72
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Andrologia (publishedVersion)
- PubMed (publishedVersion)
- 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
- congenital adrenal hyperplasia, 21-hydroxylase deficiency, 11beta-hydroxylase deficiency, azoospermia, impaired spermatogenesis, testicular function, 17-hydroxyprogesterone, testosterone, semen quality, young men, prevalence, glucocorticoid therapy
- MeSH terms
- Adrenal Hyperplasia, Congenital, Adrenal Rest Tumor, Adult, Androstenedione, Follicle Stimulating Hormone, Humans, Inhibins, Luteinizing Hormone, Male, Neoplasms, Multiple Primary, Oligospermia, Prospective Studies, Sperm Count, Sperm Motility, Testicular Neoplasms, Testosterone, Cohort Studies, Prevalence, 17-alpha-Hydroxyprogesterone, Asthenozoospermia, Azoospermia, Young Adult
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