Abstract
We read with interest the article by Chung et al. published in a recent issue of Thyroid (1). Thyroid metastases are not uncommon, with a prevalence between 1% and 24% depending on the autopsy series. The most common malignancies that metastasize to the thyroid gland are renal cell, colorectal, lung, and breast carcinomas.
Uterine sarcomas are rare, accounting for 3–7% of all uterine malignancies, and are aggressive tumors with a high metastatic potential (2). They are a heterogeneous group consisting mainly of leiomyosarcomas, endometrial stromal sarcomas, adenosarcomas, and undifferentiated endometrial sarcomas.
There have been only four previous publications on thyroid metastases of leiomyosarcomas (3 –6). We describe here a patient presenting with a synchronous intrathyroid metastasis from an undifferentiated endometrial sarcoma.
Case Report
A 58-year-old woman was referred for postmenopausal vaginal bleeding. Clinical examination revealed a large pelvic mass associated with the uterus, a firm multinodular thyroid goiter that was mobile on swallowing, and enlarged cervical lymph nodes. She had a history of a uterine leiomyoma and had undergone bilateral salpingo-oophorectomy without radiation for a benign disorder.
A pelvic magnetic resonance imaging (MRI) scan showed a 12 cm×8 cm×5 cm uterine tumor with a heterogeneous appearance involving the cervix. Endometrial and cervical biopsies were performed, and pathological examination was consistent with a highly proliferative undifferentiated carcinoma.
Thyroid ultrasonography revealed a heterogeneous nodule in the right lobe with a diameter of 3.8 cm. It was poorly defined, and showed a central calcification and intranodular vascularization. A cervical computed tomography (CT) scan revealed involvement of the isthmus, the contralateral lobe, and the larynx, and there were enlarged loco-regional lymph nodes (Supplementary Fig. S1; Supplementary Data are available at
A positron emission tomography (PET) scan revealed an abnormal uptake in the uterine mass, the thyroid nodule (Supplementary Fig. S1), the right adrenal gland, cervical and right common iliac lymph nodes, and the third thoracic vertebra. Adrenal and thyroid blood tests were normal (thyrotropin (TSH) 1.2 μU/mL, FT4 13.3 pg/mL, calcitonin <3 pg/mL).
Metrorrhagia led to significant blood loss, and total hysterectomy was performed revealing an enlarged 17 cm×13 cm×12 cm uterus (1.312 kg) infiltrated by a tumor with areas of hemorrhage and necrosis. Microscopically, the lesion was found to be a sarcomatous proliferation of the whole cervix and body of the uterus, composed of large cells with cytonuclear atypia. It had a high mitotic index and several foci of tumoral necrosis and vascular invasion (Supplementary Fig. S2). Immunohistochemical analysis confirmed the presence of an undifferentiated uterine sarcoma, as the tumor cells were negative for caldesmon and epithelial markers, but positive for CD10. Based on this, a uterine sarcoma with thyroid, adrenal, and bone metastases was diagnosed, and the patient received systemic combination chemotherapy with ifosfamide and doxorubicin.
Symptomatic tracheal compression developed and was treated by tracheotomy and cervical external beam radiation therapy. The pathological examination of the thyroid biopsies performed during tracheotomy confirmed the same characteristics as in the primary uterine tumor (CD10+, CD99+, negative cytokeratins; Supplementary Fig. S2). The patient's condition rapidly deteriorated, and she developed liver and retroperitoneal metastases and cachexia. She died two and a half months after the initial diagnosis.
Discussion
To the best of our knowledge, there have been only four previous publications on thyroid metastases from uterine leiomyosarcomas (Supplementary Table S1). We report a 58-year-old woman with thyroid metastasis from an undifferentiated endometrial sarcoma.
Clinically, the patient presented a nodular thyroid goiter and a large pelvic mass corresponding to a uterine sarcoma. She had no personal or family history of thyroid disease. The thyroid gland volume had recently increased, causing tracheal compression. Her thyroid function was normal. In the previously published cases, thyroid metastases were diagnosed between three months and four years after initial diagnosis.
Malignant disease was suspected based on the cervical CT scan and the thyroid ultrasonography. FNAB of the thyroid nodule with immunohistochemical analysis was consistent with malignancy, and was subsequently confirmed by histological analysis of surgical biopsies. The PET scan revealed associated metastases in the cervical nodes, the adrenal glands, and the bone.
This unusual presentation led to a fatal outcome two and a half months after diagnosis. Previous cases reported by Leath et al. (3) and Eloy et al. (6) followed a similar course, attesting the poor prognosis of the disease.
Conclusion
Synchronous thyroid metastasis of undifferentiated endometrial sarcoma has a very poor prognosis and a rapidly fatal outcome.
Footnotes
Author Disclosure Statement
The authors declare that no competing financial interests exist.