Endocrine Abstracts

Introduction: Granulosa cell tumors account for ~ 2–3% of all ovarian malignancies. There are two types: adult granulosa cell tumor (AGCT) and juvenile granulosa cell tumor (JGCT). JGCTs are rare and constitute 5% of all granulosa tumors. Androgen production by a JGCT is even rare and may produce virilization in patients. Thyroid abnormalities in female patients with androgen-secreting JGCT has not been documented before.

Objective:: We describe a unique case of a 14 year pubertal Caucasian girl presenting with hoarseness, hirsutism, irregular menstrual cycles and a large multi-nodular goiter with three large complex solid-cystic nodules measuring >2 cm.

Results: Thyroid function was normal and thyroid antibodies were negative. Thyroid nodule FNAC showed benign follicular changes. Testosterone level was 488 ng/dl and androstenedione levels were elevated. Karyotype, α fetoprotein and beta HCG were normal. LH was normal but FSH and estradiol levels were suppressed. Computed tomography revealed a dense, cystic solid, heterogeneous 14 cm left ovarian mass. Unilateral oophorectomy was performed. Histopathology showed intermediate grade juvenile granulosa cell tumor positive for inhibin, calretinin and negative for thyroglobulin, TTF1, PAX8 and aromatase. Testosterone (13 ng/dl), inhibin and androstenedione levels dramatically decreased immediately after surgery; LH and FSH normalized, with return of normal menstrual cycles. TSH steadily increased as FT₄ dropped over the next few months. Thyroid US showed reduction of thyroid size and nodule size by 30%. Patient was treated with L-thyroxine for hypothyroid symptoms and was stable 6 months after surgery.

Conclusion: This is the first pediatric case in the literature of ovarian androgen secreting JGCT associated with multiple large thyroid nodules. The effect of testosterone withdrawal leading to a reduced thyroid size, low FT4 with elevated TSH, has never been described before. We feel that thyroid function needs to be checked in female patients with high testosterone levels, especially after normalization of testosterone. Larger studies need to be done to look at this relationship in detail.

Declaration of interest: The authors declare that there is no conflict of interest that could be perceived as prejudicing the impartiality of the research project.

Funding: This research did not receive any specific grant from any funding agency in the public, commercial or not-for-profit sector.