Paediatric Virilizing Ovarian Tumors: A Systematic Review.
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OBJECTIVE: Virilizing ovarian tumors (VOT) are rare in the paediatric population. The literature regarding their clinical spectrum, hormonal profile, imaging characteristics, histology, and outcomes is limited. Here, we perform a systematic review on the characteristics of paediatric VOT. DESIGN AND METHODS: A systematic review was conducted in accordance with PRISMA guidelines. Databases including PubMed and Google Scholar were searched to identify English-language reports of paediatric (< 20 years) VOT with biochemical evidence of hyperandrogenemia and histological confirmation of an ovarian neoplasm. Data on demographics, clinical features, hormone profiles, imaging, tumor histology, management and outcomes were extracted and analysed. RESULTS: A total of 117 paediatric patients (median age: 15 years) with VOT were included. The most common symptoms were hirsutism (73.5%), clitoromegaly (68.4%), menstrual irregularity (n = 79), and voice change (60.7%). Children < 8 years had shorter diagnostic latency with less frequent hyperandrogenic manifestations. Baseline 17-hydroxyprogesterone (17-OHP) and dehydroepiandrosteroe sulfate (DHEAS) were elevated in 70% and 18.3% of patients, respectively. Median tumor size was 6.6 cm, whereas tumors were bilateral in eight patients. Juvenile granulosa cell tumors and Sertoli-Leydig cell tumors were most common in the < 8 years and > 13 years groups, respectively. Recurrence was rare over a median follow-up of 1 year. CONCLUSION: Hyperandrogenic manifestations are less frequent in children < 8 years with VOT. Elevated 17-OHP and/or DHEAS should not exclude the possibility of VOT. Etiological spectrum of paediatric VOT is relatively age-specific. Larger multicenter studies with longer follow-up are warranted to better delineate prognosis and management strategies.