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Submitted on February 21, 2007
Accepted on June 18, 2007
Departments of Paediatric Endocrinology, Chemical Endocrinology, Urology and Endocrinology; Radboud University Nijmegen Medical Centre, The Netherlands; Department of Urology, VU University Medical Centre, Amsterdam, The Netherlands
* To whom correspondence should be addressed. E-mail: H.Claahsen{at}cukz.umcn.nl.
Context: In male patients with congenital adrenal hyperplasia (CAH) testicular adrenal rest tumours (TART) are frequently found which may interfere with gonadal function.
Objective: Determining steroid producing features of TART
Design: Descriptive
Setting: University Medical Centre
Patients: Eight adult CAH patients with bilateral TART treated with testis-sparing surgery.
Interventions: In all but one patient spermatic veins were canulated during surgery and blood samples collected to measure the adrenal specific steroid 21-deoxycortisol (21DF) and 17-hydroxyprogesterone (17OHP) and androstenedione (A). Same parameters were measured in simultaneously taken peripheral blood. mRNA concentrations of adrenal specific enzymes CYP11B1 and CYP11B2 and ACTH and angiotensin II (AII) receptors were measured in tumours tissue.
Main outcome measures: Adrenal specific steroids/enzymes.
Results: 21DF, 17OHP and A levels were measurable in all spermatic vein samples. The ratio (mean ± SD) between spermatic vein and simultaneously taken peripheral blood samples was 37.8 ± 56.3 (21DF), 132.0 ± 249 (17OHP) and 57.0 ± 68.2 (A). CYP11B1, CYP11B2 and ACTH and AII receptor mRNAs were detected in all tumours with a strong correlation between ACTH receptor mRNA in tumours and 21DF (r = 0.85; p = 0.015), 17OHP (r = 1; p = 0.01) and A (r = 0.89; p = 0.007) concentrations in peripheral blood.
Conclusion: TART produce adrenal specific steroids and express adrenal specific enzymes and ACTH and AII receptors confirming the strong resemblance with adrenal tissue. As AII receptors are present in tumour tissue it can be hypothesized that AII may be an additional factor responsible for TART growth.
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