K.E.M. Radiology

Introduction:

Hyperandrogenism is one of the most common endocrine disorders, affecting approximately 7% of women [1]. Although polycystic ovary syndrome (PCOS) accounts for the majority of cases, it remains a diagnosis of exclusion, and ovarian and adrenal pathology must be formally ruled out [2]. Typically, total testosterone concentrations markedly above the normal range or a progressively rising trend raise concern for an autonomous androgen-secreting neoplasm [3]. Ovarian Leydig cell tumours, classified under sex cord-stromal tumours, are rare testosterone-producing neoplasms that account for fewer than 0.1–0.2% of all ovarian tumours [4]. They are characteristically small, unilateral, making them frequently occult on transvaginal ultrasound Examination (TVS) and cross-sectional imaging [5].

Ovarian vein sampling (OVS) is an interventional radiologic procedure in which blood is obtained from both ovarian veins and compared against a peripheral venous reference sample. A lateralizing gradient in testosterone concentration identifies the source ovary and guides targeted unilateral surgical resection, thereby avoiding premature menopause and loss of fertility from empirical bilateral oophorectomy [3, 6]. This is of particular relevance in women of reproductive age. A consensus on interpretation of OVS results has not been established due to the low procedurevolume and low success rates for catheterization of both ovarian veins, that has been reported at about 27–45% [7, 8].

Case presentationm

A 35-year-old nulligravida was referred for evaluation of primary infertility, irregular menstrual cycles, and hirsutism. Her symptoms had begun several years prior, and she had been initially evaluated in 2019, at which time a serum total testosterone of 1.5 μg/L (reference range: 0.20–0.80 μg/L) was documented. Pelvic ultrasound at that time had demonstrated mild bulky ovaries; a provisional diagnosis of polycystic ovarian syndrome was made, and the patient was placed on follow-up.

On subsequent re-evaluation in March 2026, serum total testosterone had risen to 4.39 μg/L — representing a progressive upward trend over seven years and a concentration approximately 5.5 times the upper limit of the reference range. Serum DHEA-S was 1540 μg/L (reference range: <5270 μg/L), well within normal limits (Table 1). 

Interventional Technique

The procedure was performed undermonitored anaesthesia care .

Right common femoral vein access was taken using a 6Fr sheath under ultrasound guidance. Initial inferior venacavography was performed, at which point the duplicated left IVC was identified (Figure 4).     

For left ovarian vein sampling, a 4Fr cobra catheter was advanced from the right IVC and navigated across to the duplicated left-sided IVC, from which the left ovarian vein was selectively catheterized using a microcatheter (Figure 5)

The right ovarian vein was cannulated using a 4Fr Sim-1 catheter and microcatheter. (Figure 6)

Samples were collected and sent for laboratory investigation.

The patient tolerated the procedure well with no immediate post-procedure complications.

5. Ovarian Vein Sampling Results: 

Selective bilateral ovarian vein sampling had been performed with simultaneous peripheral venous sampling for reference. Steroid hormone concentrations were measured by liquid chromatography–tandem mass spectrometry (LC-MS/MS), the gold-standard assay for steroid quantification owing to its superior sensitivity and specificity relative to immunoassays. Total testosterone and DHEA-S concentrations from the left ovarian vein, right ovarian vein, and peripheral sample are summarized in Table 2.

Table 2: Ovarian vein sampling results (LC-MS/MS). All concentrations expressed in μg/L (1 μg/L = 1 ng/mL).

Discussion

This case highlights two clinically important aspects of the investigation of hyperandrogenism: the diagnostic algorithm for an imaging-discordant virilizing ovarian tumour in a young woman with a prolonged history of apparent PCOS, and the technical challenge of OVS in the setting of a duplicated IVC - a previously unreported combination in the published literature.

The imaging discordance in this case -TVS suggesting a right ovarian lesion, MRI demonstrating a bulky left ovary without a discrete mass - illustrates the inherent limitations of anatomical imaging for these tumours. Leydig cell tumours may be isoechoic on ultrasonography is and below the resolution threshold of MRI, particularly at sizes below 1 cm. It has been previously demonstrated that standard imaging modalities fail to identify ovarian Leydig cell tumours in a substantial proportion of cases [3, 9]. The discordant findings in this patient precluded reliable surgical planning based on the imaging performed and OVS was considered essential prior to intervention.

The incidental finding of a duplicated IVC introduced a significant technical challenge to OVS. Duplicated IVC results from persistence of the left supracardinal vein, which normally regresses during embryological development [10]. Awareness of anomalous venous anatomy, such as duplicated IVC, is essential for procedural planning and successful bilateral catheterization.

A consensus on interpretation of OVS results for hyperandrogenism has not yet been established, owing to the limited number of published cases and low bilateral catheterization success rates [7, 8]. Moltz et al. proposed that a unilateral ovarian-to-peripheral testosterone gradient exceeding 2.7 ng/mL warrants surgical exploration [11]. Levens et al. demonstrated that a right-to-left ratio ≥1.44 (in the context of peripheral testosterone ≥4.5 nmol/L) identified 90% of right-sided tumours [8]. OVS should only be undertaken at centres highly specialized in venous sampling procedures, and only after failure to identify a source on standard imaging in patients with a clinical history strongly suggestive of an androgen-secreting tumour [9].

In the present case, the sampling results satisfied both proposed criteria for lateralization. The right ovarian-to-peripheral testosterone gradient of 148.96 ng/mL exceeded the Moltz threshold by nearly two orders of magnitude, and the right-to-left ovarian vein ratio of 32.8 far surpassed the discriminatory ratio of 1.44 described by Levens et al. The near-identical DHEA-S concentrations across the left ovarian, right ovarian, and peripheral samples (ratios approximately 0.9) excluded an adrenal contribution and reinforced a gonadal origin of the androgen excess. This biochemical lateralization to the right ovary was concordant with the equivocal right ovarian lesion seen on TVS and resolved the diagnostic uncertainty created by the discordant MRI, thereby permitting confident planning of a targeted unilateral right oophorectomy and preservation of the contralateral gonad.

References

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