Mp13-18 the Bitter Truth: Testicular Adrenal Rest Tissue (Tart) in Congenital Adrenal Hyperplasia (Cah) and Infertility

You have accessJournal of UrologyInfertility: Epidemiology & Evaluation I (MP13)1 May 2024MP13-18 THE BITTER TRUTH: TESTICULAR ADRENAL REST TISSUE (TART) IN CONGENITAL ADRENAL HYPERPLASIA (CAH) AND INFERTILITY Piotr Sluzar, Ibrahim Samy, Calum Clark, Paul Carroll, and Tet Yap Piotr SluzarPiotr Sluzar , Ibrahim SamyIbrahim Samy , Calum ClarkCalum Clark , Paul CarrollPaul Carroll , and Tet YapTet Yap View All Author Informationhttps://doi.org/10.1097/01.JU.0001008832.14212.d6.18AboutPDF ToolsAdd to favoritesDownload CitationsTrack CitationsPermissionsReprints ShareFacebookLinked InTwitterEmail Abstract INTRODUCTION AND OBJECTIVE: Testicular adrenal rest tissue (TART) is a benign growth found within testicular tissue in up to 40% of patients with congenital adrenal hyperplasia (CAH). In males with CAH, azoospermia secondary to TART is the leading cause of infertility. There is a paucity of literature on fertility optimisation in this cohort. This study aimed to provide single-centre andrology outcomes for 5 patients with CAH and TART. METHODS: A local database of 111 patients with CAH under care between January 2013 and October 2023 was retrospectively screened for diagnoses of TART in adult males. The main outcomes measured were serum hormone concentrations, semen parameters, imaging of the testes and fertility treatment success. RESULTS: In the male CAH cohort, TART was present in 5/30 patients. The average age at diagnosis was 29 (range 20-45). Prednisolone and fludrocortisone were used in 3/5 patients with alternative regimens used in 2 men. The average ACTH was 553 pg/ml (range 37-1098), testosterone was 7.97 nmol/L (range 5.3-11.6), FSH was 5.57 IU/L (range 2.2-11.9) and LH was 4.77 IU/L (range 1.8-8.8). In 2/5 patients the growth was palpable on examination and all diagnoses were confirmed on US. In all patients, the lesions were bilateral with an average testicular volume of 12.1ml (range 3.9-32.0). All masses were hypoechogenic and 4/5 showed increased vascularity, both typical features of TART. In 5/5 patients the tumour markers were negative. One patient with borderline normal semen analysis underwent 2 IVF rounds with 2 miscarriages. In a male with the lowest ACTH levels at diagnosis (37 pg/ml), a complete regression of TART with gross testicular atrophy was observed after 8 years of follow-up. The patient remained azoospermic. In the youngest male in the cohort, TART had been identified during transitional care from paediatric to adult services. The patient is undergoing sperm cryopreservation at the time of the review. CONCLUSIONS: TART is commonly associated with a reduction in fertility, particularly related to the decrease in sperm density. There is an element of obstructive azoospermia but endocrine consequences of poorly controlled CAH are also likely to be relevant. Comprehensive structured management of these patients including optimisation of endocrine status and surgical treatment including microTESE and early sperm cryopreservation are appropriate interventions. It is important to distinguish TART from Leydig cell tumour, especially in unilateral testicular lesions. Despite the literature often referring to TART as "tumours", careful counselling around the diagnosis of a benign lesion and its outcomes is required in patients transitioning from paediatric to adult services. Questions remain as to the optimal management of fertility in CAH patients with TART. Source of Funding: N/A © 2024 by American Urological Association Education and Research, Inc.FiguresReferencesRelatedDetails Volume 211Issue 5SMay 2024Page: e217 Advertisement Copyright & Permissions© 2024 by American Urological Association Education and Research, Inc.Metrics Author Information Piotr Sluzar More articles by this author Ibrahim Samy More articles by this author Calum Clark More articles by this author Paul Carroll More articles by this author Tet Yap More articles by this author Expand All Advertisement PDF downloadLoading ...