Ovarian steroid cell tumor (not otherwise specified) with subsequent spontaneous pregnancy after tumor removal: a case report and literature review

Figures & data

Figure 1. Findings of virilization. (a) Hirsutism of face. (b) Male-pattern baldness (with permission from the patient).

Figure 2. (a) Ultrasonographic appearance of left ovarian hyperechoic mass sized 5 × 5 cm. (b) Computed tomography of abdomen shows an enhancing lesion (7.8 × 5.2 × 4.8 cm) with contained 5.8 × 4.7 × 4.5-cm well-defined enhancing fatty mass without calcification likely arising from the left ovary (arrow).

Table 1. Body weight and laboratory results before and after surgery.

	Before surgery	1 month after surgery	5 months after surgery	Reference range
Body weight (kg)	90		72
Cortisol at 8 AM (μg/dL)	12.27			5.9-26.10
Total testosterone (ng/dL)	207	48		20-80
17α-hydroxyprogesterone (ng/dL)	1,087	154
Estradiol (pg/mL)	59.72	78.97
Follicle stimulating hormone (mIU/mL)	3.7	2.47
Luteinizing hormone (mIU/mL)	3.94
Free thyroxine (ng/dL)	1.35			0.7-1.75
Thyroid stimulating hormone (mIU/L)	3.4	3.4		0.25-4.00
Prolactin (ng/mL)	18.08			3.6-18.9
Sodium			137.8	136-145
Potassium			4.17	3.4-4.5
Bicarbonate			24.3	22-29
Chloride			100.7	98-107
Fasting blood sugar (mg%)	89		81	74-99
HbA1C (%)	5.1		5.1	4.8-5.9

Figure 3. (a) Cross-sectional appearance of the left ovary. The ovarian tumor cut surfaces are well-circumscribed, yellowish color, and lobulated. (b) Tumor cells have round to polygonal shape with eosinophilic to clear cytoplasm and oval nucleus. (c) Tumor cells arrange in cord and sheet (*) with fibrovascular tissue (arrow). (d) Tumor cells show positive for alpha-inhibin immunostaining.

Table 2. A summary of steroid cell tumors, not otherwise specified with spontaneous pregnancy reported after surgery.

	Sielert et al. 2013 [2]	Uyanıkoglu et al. 2020 [3]	Our case
Age (years)	20	34	31
Presenting symptoms	Amenorrhea, hirsutism, acne, deepening voice	Amenorrhea, hirsutism, deepening voice, alopecia; had been diagnosed with PCOS and CAH	Amenorrhea, hirsutism, alopecia
Duration of infertility	No fertility problems	2 years, history of failed 1 IVF cycle	10 years
Hormonal profile	T: 485 ng/dL A: 1738 ng/dL DHEA: 1459 ng/dL	T: 416 ng/dL 17-OHP: 2000 ng/mL DHEA-S: 385.9 μg/dL	T: 207 ng/dL 17-OHP: 1087 ng/dL E: 59.72 pg/mL Cortisol: 12.27 μg/dL
Tumor size	5.4 × 4.5 cm	4.2 × 3.2 cm	6.0 × 6.0 cm
Evidence of malignancy	No	No	No
Resumption of menses after surgery	4 months	n/a	1 month
Time to pregnancy after surgery	9 months; reported condom breakage and failed LNG 1.5 mg orally	4 months	12 months
Pregnancy outcome	Uncomplicated pregnancy; female infant, 3,579 g at 40 weeks gestation	Healthy infant at term	Uncomplicated pregnancy; male infant, 3,495 g at 38 weeks gestation

T: total testosterone, 17-OHP: 17α-hydroxyprogesterone, A: androstenedione, DHEA: dehydroepiandrosterone, DHEA-S: dehydroepiandrosterone sulfate, E: estradiol.

LNG: levonorgestrel, PCOS: polycystic ovarian syndrome, CAH: congenital adrenal hyperplasia.

Sielert L, Liu C, Nagarathinam R, et al. Androgen-producing steroid cell ovarian tumor in a young woman and subsequent spontaneous pregnancy. J Assist Reprod Genet. 2013;30(9):1157–1160.

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Uyanıkoglu H, Ozer G, Kahraman S. A spontaneous pregnancy and live birth in a woman with primary infertility following the excision of an ovarian adrenal rest tumor: a rare case. Clin Exp Reprod Med. 2020;47(4):319–322.

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