Testicular adrenal rest tumors in children with congenital adrenal hyperplasia

Waad M. Al-Ghamdi; Mohamed A. Shazly; Abdulmoein E. Al-Agha

doi:10.15537/smj.2021.42.9.20210257

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Figure 1
- Bilateral testicular involvement with hypoechoic TART in patient number one.
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Figure 2
- Hyperechoic testicular adrenal rest tumor (TART) on the right testes of patient number 2.

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Table 1

- Characteristic features of patients with testicular adrenal rest tumors (TARTs) including tanner staging, testicular volume, and penile length.

Patient number	Age at TART detection (years)	*Presence of signs and symptoms	Hyperpigmentation	Palpable testicular mass	Tanner stage “testes”	Tanner stage “pubic hair”	Testicular volume (mm)	Penile length (cm)
1	16.6	yes	Yes	No	IV	V	R:15 L:15	17
2	20	no	No	Yes, nodular	V	V	R:20 L:25	17
3	10.8	no	No	No	I	I	R:3 L:2.5	6
4	15.6	no	Yes	No	IV	IV	R:20 L:20	9
5	9.4	no	Yes	No	I	II	R:3 L:3	6
6	8.9	yes	Yes	No	II/III	III	R: 8 L: 10	9

↵* presence of scalp alopecia, acne, or obesity

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Table 2

- Radiological investigation including sonographic features of testicular ultrasound in patients with congenital adrenal hyperplasia with testicular adrenal rest tumors (TARTs), including bone age advancement.

Patient number	TART largest diameter on ultrasonography (cm)		Bilateral	Echogenicity	Vascular	Ill-defined border	Site	Bone age advancement
Patient number	R	L	Bilateral	Echogenicity	Vascular	Ill-defined border	Site	Bone age advancement
1	Largest: 0.4x0.4		yes	Hypoechoic	increased	yes	-	-
2	3x1	1.5x1.3	yes	Eccentric hyperechoic	increased	-	-	-
3	0.2x0.4	0.3x0.3	yes	Hypoechoic	Non-vascular	yes	-	by 1.3 years
4	0.4x0.3		no (right)	Hypoechoic	increased	-	mediastinum	by 4. 8 years
5	0.7x0.6	0.3x0.4	yes	Hypoechoic	increased	-	mediastinum	by 3.4 years
6	Multiple small		yes	Hypoechoic	increased	-	mediastinum	by 2.10 years

R: right, L: left

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Table 3

- Hormonal assays of patients with congenital adrenal hyperplasia (CAH) with testicular adrenal rest tumor (TARTs).

Patient number	ACTH (Pmol/L)		17-OHP (ng/mL)	DHEA-S (umol/L)	Testosterone (nmol/L)	LH (mIU/L)
Patient number	Prior	After	17-OHP (ng/mL)	DHEA-S (umol/L)	Testosterone (nmol/L)	LH (mIU/L)
1	170	96	1.8	0.84	34	5.68
2	28	1.11	2.7	0.889	10.5	17.4
3	117	9.79	0.43	0.19	0.87	0.2
4	80	1.96	5.9	14	20.51	4.5
5	77	1.43	3.4	0.12	0.3	1.2
6	105	10	21.7	0.31	0.91	2.01

ACTH: adrenocorticotropic hormone, 7-OHP: 17-hydroxy progesterone, DHEA-S: dehydroepiandrosterone, LH: luteinizing hormone

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Table 4

- Comparison between TART negative and positive cases as regard all parameter.

Parameters	TART								P-value**	Sig
	Negative				Positive
	Mean±SD	Median	IQR		Mean±SD	Median	IQR*
Age	12.20±5.16	11.00	8.00	15.00	12.83±4.62	12.50	9.00	15.00	0.797	NS
BMI	19.78±6.14	18.10	15.20	23.20	25.55±4.55	25.40	21.30	27.70	0.054	NS
ACTH	18.73±26.00	9.00	2.20	21.10	96.17±47.41	92.50	77.00	117.00	0.001	HS
Testosterone	5.07±7.27	0.35	0.09	11.00	11.18±13.70	5.71	0.87	20.50	0.161	NS
DHEA-S	1.75±3.81	0.15	0.00	1.41	2.72±5.53	0.58	0.19	0.88	0.274	NS
17-OHP	6.72±9.01	2.80	0.44	14.80	5.99±7.91	3.05	1.80	5.90	0.697	NS
LH	4.36±10.37	0.20	0.00	4.50	5.17±6.34	3.26	1.20	5.68	0.114	NS
FSH	12.91±37.86	0.20	0.00	3.67	5.72±7.46	3.09	0.70	6.73	0.069	NS

TART: testicular adrenal rest tumor, Sig: significance level, IQR: Interquartile range, BMI: body mass index, ACTH: adrenocorticotropic hormone, DHEA-S: dehydroepiandrosterone, 17-OHP: 17-hydroxy progesterone, LH: luteinizing hormone, FSH: follicle stimulating hormone