Precocious Pseudo-Puberty with Testicular Enlargement: Two Cases of Leydig Cell Tumor with Different Histopathological Results

Figures & data

Figure 1 Acne and facial hair (A) and enlarged penis with scant pubic hair (B) in a 6-year-old boy, showing signs of precocious pseudo-puberty as shown on both cases. Advanced bone age of Case 2 (C).

Table 1 Characteristics of Patients

Age at Diagnosis	Physical Examination	Imaging
6 years old	BW 33.5 kg (>97th percentile), height 141 cm (>97th percentile). Enlarged left testicle, 3 x 3 cm in size, with firm nodule, no tenderness. Right testicle 1 x 1 cm in size. Masculine habitus with acnes and facial hair. The pubertal state of the patient was A1P3G3–4.	US: Left testicle, 2.65 x 2.54 x 3.0 cm in size with peripheral and central vascularization, mostly occupied by heterogenous parenchymal lesion. Normal right testis, 0.79 x 0.92 x 1.3 cm in size
6 years old	BW 40 kg (>97th percentile), height 136 cm (>97th percentile). Left testicle was enlarged (10 mL), painless, without signs of inflammation. Right testicle was slightly enlarged (5 mL) and soft. The pubertal state was A1P3G3. Pubertal features, such as pubic hair, acne, and fair facial hair were observed.	US: Heterogeneous, hypoechoic mass (1.6 x 1.3 cm) with calcification in the center of the testis was shown on the left side X-ray: bone age was significantly advanced at 12–13 years. MRI: enlarged left testicle (1.98 x 2.12 x 2.61 cm) with bilateral inguinal lymphadenopathy. No enlargement on adrenals nor any metastatic features in other organs.

Abbreviation: BW, bodyweight.

Figure 2 Ultrasound of the testicles of Case 1 (A) and Case 2 (B).

Table 2 Hormonal Assessment of the Patients

	Patient 1 Pre-Operative	Patient 1 6 Months after Surgery	Patient 2 Pre-Operative	Patient 2 9 Months after Surgery	Reference Level
LH (mIU/mL)	0.1 (L)	1.8 (H)	0.83 (N)	3.43 (H)	0.7–1.2
FSH (mIU/mL)	0.1 (L)	2.01 (N)	<0.1 (L)	7.25 (H)	0.16–3.5
Testosterone (ng/dL)	387.6 (↑15x ULN)	240.3 (↑9x ULN)	281.35 (↑11x ULN)	30 (↑1,2 x ULN)	≤25
AFP (ng/mL)	0.85 (L)	2.0 (N)	0.68 (N)	0.45 (N)	0.68–8.78
β-HCG (mIU/mL)	<1.2 (N)	<1.2 (N)	<1.2 (N)	-	<5
LDH (U/L)	160 (N)	-	295 (N)	-	160–360

Abbreviations: L, low; N, normal; ULN, upper limit of normal.

Figure 3 Macroscopic anatomy findings of the left testis from the patient of Case 1 (A) and Case 2 (B). Note a well circumscribed yellowish-brownish mass inside the testis.

Figure 4 (A) The tumor mass was lobulated had indistinct border; (B) spherical nuclei of tumor cells, pleomorphic, granulated eosinophilic cytoplasm, (high power field, 400x); (C) Reinke crystal. (D) Solid tumors formed nest.