Endocrine Abstracts

Introduction: Testicular adrenal rest tumours (TART) are benign corticotrophin-dependent tumours that occur in males with congenital adrenal hyperplasia (CAH). We present a patient with bilateral large TART as a consequence of poor compliance to treatment and follow-up for his CAH.

Case: A 25-year-old gentleman presented to the endocrine clinic in 2009 with a history of tiredness, reduced libido and bilateral large testicles, which he wanted surgically removed. He had been diagnosed with CAH during the antenatal period and commenced on steroid replacement therapy soon after birth. However, he stopped taking steroid replacement in 2000 because of domestic/family issues and although his testicles were felt to be lumpy in 2002, further follow-up and investigation was difficult. On examination he was well with normal secondary sexual features, but his testicles felt hard and three times the normal size.

Investigations: His biochemical test results are shown in the Table 1. An ultrasound confirmed large testicles. He was commenced on steroid replacement therapy and referred for orchidectomy and replacement prosthesis. Both testicles measured 8.5×5×4 cm in size with no recognisable testicular parenchyma (stage 5): the histology was characteristic of TART. His serum testosterone levels fell to the lower limit of the normal range post-surgery.

Conclusion: Although not malignant, TART can result in irreversible damage of testicular tissue and infertility. Treatment with glucocorticoid replacement therapy can stabilise or repress growth of these tumours. This case highlights the importance of compliance to treatment and follow-up to prevent testicular damage as a result of TART in patients with CAH.