Sitasuwan Tullaya, Hanamornroongruang Suchanan, Peerapatdit Thavatchai, Thongtang Nuntakorn
Division of Endocrinology and Metabolism, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, 10700, Thailand.
Department of Pathology, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, 10700, Thailand.
BMC Endocr Disord. 2015 Nov 4;15:68. doi: 10.1186/s12902-015-0060-z.
Coexisting of Graves' disease and functioning struma ovarii is a rare condition. Although the histology of struma ovarii predominantly composed of thyrocytes, the majority of the patients did not have thyrotoxicosis. The mechanism underlying the functioning status of the tumor is still unclear but the presence of thyroid stimulating hormone receptor (TSHR) is thought to play a role. Here we describe the patient presentation and report the TSHR expression of the tumor.
A 56-year old Asian woman presented with long standing thyrotoxicosis for 23 years. She was diagnosed with Graves' disease and thyroid nodules. She had bilateral exophthalmos and had high titer of plasma TSHR antibody. Total thyroidectomy was performed and the histologic findings confirmed the clinical diagnosis. The patient had persistent thyrotoxicosis postoperatively. Thyroid uptake demonstrated the adequacy of the thyroid surgery and the whole body scan confirmed the presence of functioning thyroid tissue at pelvic area. The surgery was scheduled and the patient had hypothyroidism after the surgery. The pathological diagnosis was struma ovarii at right ovary. We performed TSHR staining in both the patient's struma ovarii and in 3 cases of non-functioning struma ovarii. The staining results were all positive and the intensity of the TSHR staining of functioning struma ovarii was the same as that in other cases of non-functioning tumors, suggesting that the determinant of functioning struma ovarii might be the presence of TSHR stimuli rather than the intensity of the TSHR in the ovarian tissue.
In patients with Graves' disease with persistent or recurrent thyrotoxicosis after adequate ablative treatment, the possibility of ectopic thyroid hormone production should be considered. TSHR expression is found in patients with functioning and non-functioning struma ovarii and cannot solely be used to determine the functioning status of the tumor.
格雷夫斯病与功能性卵巢甲状腺肿并存是一种罕见的情况。尽管卵巢甲状腺肿的组织学主要由甲状腺细胞组成，但大多数患者并无甲状腺毒症。肿瘤功能状态的潜在机制仍不清楚，但甲状腺刺激激素受体（TSHR）的存在被认为发挥了作用。在此，我们描述该患者的表现并报告肿瘤的TSHR表达情况。
一名56岁的亚洲女性因长期甲状腺毒症23年就诊。她被诊断为格雷夫斯病和甲状腺结节。她有双侧突眼且血浆TSHR抗体滴度高。进行了全甲状腺切除术，组织学检查结果证实了临床诊断。患者术后仍有持续性甲状腺毒症。甲状腺摄取显示甲状腺手术充分，全身扫描证实盆腔区域存在功能性甲状腺组织。安排了手术，术后患者出现甲状腺功能减退。病理诊断为右侧卵巢甲状腺肿。我们对该患者的卵巢甲状腺肿以及3例无功能性卵巢甲状腺肿进行了TSHR染色。染色结果均为阳性，功能性卵巢甲状腺肿的TSHR染色强度与其他无功能性肿瘤病例相同，这表明功能性卵巢甲状腺肿的决定因素可能是TSHR刺激的存在而非卵巢组织中TSHR的强度。
对于接受充分消融治疗后仍有持续性或复发性甲状腺毒症的格雷夫斯病患者，应考虑异位甲状腺激素产生的可能性。在功能性和无功能性卵巢甲状腺肿患者中均发现了TSHR表达，且不能仅用其来确定肿瘤的功能状态。
