November 08, 2013
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Testicular adrenal rest tumors not linked to poor endocrine control

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Poor disease control in patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency did not appear to play a role in development of testicular adrenal rest tumors, according to results of a retrospective longitudinal study.

“The adrenal-like features of [testicular adrenal rest tumors] are a major argument for the hypothesis that [testicular adrenal rest tumors] originate from aberrant adrenal cells within the gonads,” researchers wrote. “Poor quality of disease control has been advocated to be one of the main predictive factors for [testicular adrenal rest tumor] development.”

To investigate this potential connection, the researchers evaluated 50 men with classic 21-hydroxylase deficiency, 31 of whom were classified as salt wasting and 19 of whom were classified as simple virilizing. Long-term parameters of endocrine control during childhood and adolescence, including 24-hour urine pregnanetriol, serum 17-hydroxyprogesterone, onset and stage of pubic hair development, testicular growth and bone age in relation to chronological age were assessed.

Of the 50 men included in the analysis, 28 developed testicular adrenal rest tumors (salt wasting, n=19; simple virilizing, n=9) as detected by testicular ultrasound at a median age of 27 years. Results revealed no significant difference in pregnanetriol concentrations over lifetime between patients with and without tumors (P=.35). Similarly, the researchers found no significant differences in long-term markers of disease control, including testicular volume (P=.28), bone age (P=.76) and pubic hair stage (P=.73) when comparing patients with vs. without tumors.

Additionally, BMI and impairment of final height in relation to midparental target height were similar among patients with and without tumors.

“Our longitudinal analysis demonstrates no association between [testicular adrenal rest tumor] presence and parameters of disease control,” the researchers wrote. “These data, therefore, argue for other mechanisms more relevant for [testicular adrenal rest tumor] induction including those occurring during fetal development.”

Disclosure: The researchers report no relevant financial disclosures.