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A 14-year-old female presented with intermittent abdominal pain, deepening of voice and amenorrhoea for 4 months. Twelve months earlier she had had a thyroidectomy because of familial multinodular goiter and had, subsequently, received substitution with L-thyroxine. At the time of admission, a high serum testosterone level was detected. The dexamethasone suppression test confirmed the hormonal autonomy and magnetic resonance imaging (MRI) visualized a solid tumor within the left ovary. The pathological diagnosis after left salpingo-oophorectomy was Sertoli-Leydig cell tumor. The patient has remained disease-free for 6 years. Pediatr Blood Cancer 2008;51:543–545. © 2008 Wiley-Liss, Inc.