Bilateral Testicular Adrenal Rest Tumours in a Male with Well-Controlled Congenital Adrenal Hyperplasia

Ambekar AN, Sonwane AC, Inamdar S, Reddy JP, Kiran SG, Sethi BK and Modi KD

Published on: 2025-06-07

Abstract

A man in his 20s consulted a surgeon due to painless Swelling of the scrotum for the last four to five months. He had no other symptoms and denied a history of trauma pain or breast swelling. Ultrasonography of the scrotum showed irregular, eccentric masses in both the testes. He underwent bilateral surgical excision of the masses with the suspicion of malignancy. Histopathological examination showed a well-circumscribed tumour with large polygonal cells arranged in nests with abundant eosinophilic cytoplasm. Such histological findings are encountered in Leydig cell tumour as well as testicular adrenal rest tumours (TARTs). However, the bilateral location and the presence of lipofuscin pigment favoured TARTs. The patient was referred to the endocrinology services with this report. Hormonal evaluation revealed normal serum 17-hydroxyprogesterone (17-OHP) and testosterone with mildly elevated LH. On further questioning, he admitted being on treatment for CAH and being irregular with the prescribed medication till about a month, when he started taking them regularly when his cousin remarked that intermittent use might lead to health issues. This case highlights the possible occurrence of TARTs even in seemingly well-controlled cases of CAH. He was started on prednisolone and fludrocortisone and counselled for regular follow-up.  This case illustrates the importance of thorough history taking and keeping TARTs as a differential diagnosis of testicular tumours.