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Thyrotoxic adenoma followed by atypical hyperthyroidism due to struma ovarii: Clinical and genetic studies

par Ciccarelli, Antonio;Socin, Hernan Valdes;Parma, Jasmine ;Khoo, Sok Kean;Schoumans, Jacqueline;Colao, Annamaria;Hamoir, Etienne;Beckers, Albert
Référence European journal of endocrinology, 150, 4, page (431-437)
Publication Publié, 2004-04

Article révisé par les pairs

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Résumé :

Objective: Atypical forms of hyperthyroidism represent a1 diagnostic challenge for clinicians. Struma ovarii is an ovarian teratoma and constitutes a rare cause of ectopic thyroidal hormonal production. We describe a case of struma ovarii that combined two different sources of hyperthyroidism in the same patient and report genetic studies in order to contribute a better understanding of the autonomy and tumorigenesis of the struma ovarii. Case report: A 73-year-old nulliparous woman presented a thyroid toxic adenoma that was successfully treated with 10mCi radioiodine. Unexpectedly, a new onset of hyperthyroidism prompted us to look for a second etiology. A whole-body scan with 123I detected a pelvic hyperfixation suggesting struma ovarii, and a thyroid differentiated left ovarian teratoma 3 cm in size was surgically removed. We screened for mutations of thyroid-stimulating hormone receptor and Gs-α protein genes, as these mutations are common in thyroid adenomas. We did not identify any mutations. Androgen receptor study demonstrated a monoclonal status. Comparative genomic hybridization did not reveal any chromosomal abnormality. However, loss of heterozygosity analysis showed several structural abnormalities, compared with the majority of benign ovarian teratomas, which show a normal karyotype. Conclusions: This is the first well-documented report of thyrotoxic struma ovarii revealed after treatment of a single thyroid toxic adenoma. We have shown in this case that struma ovarii originates from a single germ cell, and, albeit benign, this tumor presents several chromosomal abnormalities. Struma ovarii-induced hyperthyroidism is likely to be mediated by mechanisms different from those of the classical thyroid toxic adenoma. © 2004 Society of the European Journal of Endocrinology.