Szczepanek-Parulska Ewelina, Pioch Anna, Cyranska-Chyrek Ewa, Wolinski Kosma, Jarmołowska-Jurczyszyn Donata, Janicka-Jedynska Malgorzata, Majewski Przemyslaw, Zabel Maciej, Ruchala Marek
Department of Endocrinology, Metabolism and Internal Medicine, Poznan University of Medical Sciences, Poznan, Poland.
Department of Clinical Pathology Poznan University of Medical Sciences, Przybyszewskiego 49, 60-355 Poznan, Poland.
Folia Histochem Cytobiol. 2019;57(1):35-42. doi: 10.5603/FHC.a2019.0004. Epub 2019 Mar 29.
Struma ovarii (SO) is a monodermal teratoma in which thyroid tissue comprises more than 50% of the tumour. Papillary thyroid cancer (PTC) in SO is a rare finding, as only 5% of SO cases undergo malignant transformation. Malignant SO is usually asymptomatic and infrequently diagnosed preoperatively. Because of its rarity, there is no consensus about diagnosis and management, while treatment and follow-up procedures are not clearly established.
Herewith, we report two cases of PTC in SO. The first patient was a 25-year-old woman diagnosed with bilateral ovarian tumours. The second patient, 19-year-old woman, presented with unilateral ovarian mass. Both patients were qualified for surgical excision of the tumours. Histopathological specimens underwent both conventional histopathological assessment and immunohistochemical staining.
In the first patient histopathology revealed SO with two foci of PTC. Immunohistochemically a positive expression of CK7, CK19, p63 and thyroglobulin (Tg) confirmed the diagnosis. She underwent total thyroidectomy in 2016 in order to enable ablative radioiodine therapy and facilitate further thyroglobulin monitoring. Unfortunately, the patient was lost from follow-up. In the second patient, histopathological diagnosis was follicular variant of PTC in SO. Postoperatively, a pelvic CT revealed osteolytic lesion 6 cm in size, being a metastatic change. The patient underwent unilateral ovariectomy, total thyroidectomy and multiple cycles of radioiodine therapy. Currently, 9 years following the diagnosis, the patient achieved disease remission.
PTC in SO still remains a diagnostic and therapeutic challenge. Immunostaining for CK7, CK19, p63 and Tg might be helpful in histopathological diagnosis. The decision on the need of total thyroidectomy and radioiodine therapy should be made individually. However, thyroid remnant ablation increases the sensitivity and specificity of follow-up testing using serum Tg level as a tumour marker.
卵巢甲状腺肿（SO）是一种单胚层畸胎瘤，其中甲状腺组织占肿瘤的50%以上。SO中的甲状腺乳头状癌（PTC）是一种罕见的发现，因为只有5%的SO病例会发生恶性转化。恶性SO通常无症状，术前很少被诊断出来。由于其罕见性，关于诊断和管理尚无共识，而治疗和随访程序也未明确确立。
在此，我们报告2例SO中的PTC病例。首例患者为一名25岁女性，被诊断为双侧卵巢肿瘤。第二例患者为一名19岁女性，表现为单侧卵巢肿块。两名患者均符合手术切除肿瘤的条件。组织病理学标本进行了常规组织病理学评估和免疫组化染色。
首例患者的组织病理学检查显示为SO伴两个PTC病灶。免疫组化显示CK7、CK19、p63和甲状腺球蛋白（Tg）呈阳性表达，确诊了诊断。她于2016年接受了全甲状腺切除术，以便进行消融性放射性碘治疗并便于进一步监测甲状腺球蛋白。不幸的是，该患者失访。第二例患者的组织病理学诊断为SO中的PTC滤泡变体。术后盆腔CT显示一个6厘米大小的溶骨性病变，为转移改变。该患者接受了单侧卵巢切除术、全甲状腺切除术和多个周期的放射性碘治疗。目前，诊断9年后，该患者实现了疾病缓解。
SO中的PTC仍然是一个诊断和治疗挑战。CK7、CK19、p63和Tg的免疫染色可能有助于组织病理学诊断。是否需要进行全甲状腺切除术和放射性碘治疗应个体化决定。然而，甲状腺残余物消融可提高以血清Tg水平作为肿瘤标志物的随访检测的敏感性和特异性。
