Longitudinal Characterization and Sonographic Staging of Testicular Adrenal Rest Tumors
Authors' conclusion
TART formation in CAH is frequently found in prepubertal boys, with a characteristic pattern of development resulting in a proposed radiologic staging. Pediatric screening is recommended, especially in those with a SW phenotype/genotype.
Study context
- Population
- Males with classic congenital adrenal hyperplasia (CAH) followed prospectively from childhood to adulthood783 US evaluated
- Participants
- 36 males
- Outcome
- Age of TART onset and associated factors; sonographic natural history of TART
Methodological detailsserial scrotal US every 6 months in childhood and annually in adulthood, survival analysis and longitudinal models
- Serial scrotal US every 6 months in childhood and annually in adulthood
- Survival analysis and longitudinal models
Main finding
As reported in the abstract
Developed testicular adrenal rest tumors (TART)
27
75%
Mean age of TART onset
13.6 ± 4.9 years
Risk of TART before age 10
∼22%
Observational data: associations, not effects.
Further findings
Factors associated with TART at any given visit (multivariable analysis)
| OR (95% CI) | P value | ||
|---|---|---|---|
| SW phenotype | 6.83 (2.36–19.77) | P < 0.001 | |
| Higher hydrocortisone dose equivalents | 1.10 (1.03–1.18) | P = 0.006 | |
| Elevated ACTH | 1.0011 (1.0005–1.0017) | P < 0.01 | |
| 0.050.21520Odds ratio (log scale) |
Hydrocortisone dose equivalent · last visit
- Hydrocortisone dose equivalent: 19 patients with TART at last visit
Factors associated with TART at any given visit (multivariable analysis)
- TART presence associated with elevated ACTH, 17-hydroxyprogesterone, androstenedione and plasma renin activity, younger age at diagnosis, salt-wasting (SW) phenotype and null/In2G mutations
Sonographic progression of TART
- Characteristic progression from a single hypoechoic nodule to multiple hypoechoic nodules to a single conglomerate mass near the mediastinum testis
Original publication
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