Received Date: March 13, 2014; Accepted Date: May 20, 2014; Published Date: May 27, 2014
Citation: Ionescu L, Danila R, Blaj M, Savin M, Vulpoi C, et al. Anaplastic Thyroid Carcinoma or Thyroid Metastasis from Cholangiocarcinoma? A Case Report. Journal of Surgery [Jurnalul de chirurgie] 2015; 11(2): 381-383 DOI:10.7438/1584-9341-11-2-7
Copyright: © 2015 Ionescu L, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
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Anaplastic thyroid carcinoma presents as an extremely localy invasive neck mass while metastases in the thyroid are most commonly described as small, indolent, solitary nodules usually originating from kidney, breast, lungs and skin tumors. We report the case of a 74-year old male patient illustrating the difficulties of differential diagnosis between an anaplastic thyroid carcinoma and a thyroid metastasis of a peripheral cholangiocarcinoma in a cirrhotic patient diagnosed and operated for a locally advanced thyroid tumor. The history, clinical and imagistical features strongly pleaded for the diagnosis of anaplastic thyroid carcinoma presumably with liver metastases, also supported by the rapid recurrence following total thyroidectomy. Immunohistochemical tests showed a malignant carcinomatous proliferation with anaplastic prophile. Positive immunochemical staining for cytokeratin AE1/AE3, CK7 and negative CK20, AFP, CD15, CD30, CD5, TTF1 and thyroglobulin directed the possible diagnosis toward a secondary thyroid tumor from a peripheral cholangiocarcinoma. Immunohistochemical tests showed a malignant carcinomatous proliferation with anaplastic prophile. Positive immunochemical staining for cytokeratin AE1/AE3, CK7 and negative CK20, AFP, CD15, CD30, CD5, TTF1 and thyroglobulin directed the possible diagnosis toward a secondary thyroid tumor from a peripheral cholangiocarcinoma. A CT-guided percutaneous hepatic punction biopsy was planned but the patient presented an ischemic stroke with fatal outcome. In conclusion, in spite of surgical treatment the rapid recurrent thyroid cancer either primary or metastatic had a poor prognosis with fatal outcome mainly in the presence liver cirrhosis and cardio-vascular co-morbidities.
Metastatic tumors in the thyroid gland occur in as many as 24%of subjects when examined at autopsy and most commonly primarytumors are located in the kidney, breast, lung, and malignantmelanoma of the skin. Generally, a metastatic tumor in the thyroidgland presents as a solitary nodule that may be the initial evidence ofdisease or the first presentation of recurrent disease but more oftenthere is a widespread metastatic disease present and the manifestationsin the thyroid gland are clinically unimportant. Anaplastic carcinomadescribes an undifferentiated malignancy derived from more welldifferentiatedthyroid follicular epithelium. In contrast to the generallyindolent nature of differentiated thyroid carcinoma, anaplasticcarcinoma represents one of the most aggressive human neoplasms,with a disease-specific mortality of at least 90%. Occasionally, it may bedifficult to determine if the specimen represents metastatic disease orif it is originating from the thyroid gland, such as an anaplastic thyroidcarcinoma .
We present a case illustrating the difficulties of differential diagnosisbetween an anaplastic thyroid carcinoma with liver metastases and athyroid metastasis of a peripheral cholangiocarcinoma in a cirrhoticpatient diagnosed and operated for a locally advanced thyroid tumor.
A 74-year old male patient was referred to surgery from theendocrinology department for a thyroid tumor with compression signs.The patient was previously diagnosed with C virus liver cirrhosis, type2 diabetes mellitus, arterial hypertension, ischemic heart disease andanemia. The patient reported a 4 month history of fatigue and weight loss and more recently (2 weeks) neck pain and enlargement of theanterior cervical region with dyspnoea and disphagia. A cervical lymphnode biopsy previously performed in the ENT department revealedjust a chronic nonspecific lymphadenitis. The physical examinationshowed a large, irregular, hard and fixed tumor of the right thyroid lobewith multiple laterocervical lymphadenopathies. The lab tests showed anormal thyroid function and calcitonine level, AFP, CEA and CA19-9within normal range. Ultrasound of the thyroid described an extensivetumor of the right lobe with bilateral cervical lymphadenopathies.Thyroid scintigraphy revealed multiple areas of hypo and affixationof 99m Tc in both lobes. Computer tomography (CT) of the neckrevealed the thyroid gland almost completely replaced by a 64/89/85mm solid, inhomogeneous tumor, predominantly developed in theright lobe and deviating trachea, larynx and hypopharinx to the left.Right internal jugular vein was thrombosed without demarcation limitfrom the tumor and multiple laterocervical and superior mediastinumlymphadenopathies were present (Figure 1).
FNAC (fine needle aspiration cytology) showed a suspiciouscytology-Bethesda V. Routine preoperative laryngoscopy was normal. Under the suspicion of thyroid malignancy the patient was operated anda total thyroidectomy with lymphadenectomy of the central and lateralcompartments of the neck was performed. The frozen section examrevealed a massive malignant infiltration with marked pleomorphism.The final pathology described a massive malignant infiltration withmarked pleomorphism and large areas of necrosis (Figure 2).
Immunohistochemical tests were performed but could not distinctbetween an anaplastic carcinoma with lymphoepithelial aspect and ametastatic carcinoma from a cholangiocarcinoma (Table I).
Table I: Immune Histochemistry Tests.
|Citokeratin CK AE1/AE3, CK7||Intense Positive|
|Thyroglobulin (TG), TTF1||Negative|
|LCA||Intense Positive in peritumoral lymphoid tissue but negative in the tumoral cells|
After 2 months he was readmitted with signs of local recurrence.CT showed a 90/86/82 mm tumor mass in right thyroid space withnecrotic areas which compressed the carotid sheath, larynx, andesophagus. CT of the abdomen showed multiple nodules in the VI, VII,VIII segments of the liver, suggestive for liver metastases or peripheralcholangiocarcinoma. An exploratory laparoscopy with liver biopsy wasintended but the patient suffered an ischemic stroke with grade II coma(extensive subarahnoidian hemorrhage.) and deceased 10 ten days laterin the intensive care unit.
The biological behaviour, clinical and imagistical features shouldnormally allow an obvious distinction between an undifferentiatedprimary thyroid carcinoma and a secondary tumor in the gland.Whereas anaplastic thyroid carcinoma presents as an extremely localyinvasive neck mass, metastases in the thyroid are most commonlydescribed as small, indolent, solitary nodules. In a large series from theMayo Clinic, the average size of the thyroid metastatic nodules was 3cm . The incidence of thyroid metastases secondary to any type ofprimary tumor is reported between 1.9-9.5% and surgical resection ofthe thyroid metastasis ranges between 0.02 and 1.4% [3-8]. The survivalis poor, ranging between 1 and 12 months [9-12]. Most frequentlythe primary tumor is renal and only two cases of thyroid metastasessecondary to cholangiocarcinoma were reported on Pubmed database[3,4].
In our case, the history, clinical and imagistical featuresstrongly pleaded for the diagnosis of anaplastic thyroid carcinomapresumably with liver metastases, also supported by the rapidrecurrence following total thyroidectomy. Immunohistochemicaltests showed a malignant carcinomatous proliferation with anaplasticprophile. Positive immunochemical staining for cytokeratin AE1/AE3, CK7 and negative CK20, AFP, CD15, CD30, CD5, TTF1 and thyroglobulin raised the suspicion of a secondary thyroid tumor froma peripheral cholangiocarcinoma. This hypothesis was also supportedby the diagnosis of macronodular liver cirrhosis and presumablya neoplasm in the segment VII. Although the tumoral markers for hepatoma or cholangiocarcinoma were within normal limits,immunohistochemistry tests raised the possibility of a metastaticthyroid tumor from a cholangiocarcinoma. Unfortunately, the patientpresented a fatal stroke in the day when he was listed for a CT-guidedpercutaneous hepatic punction biopsy. This exploration would haveenabled us to delineate with accuracy the relationship between thethyroid tumor and the liver nodules.
In conclusion, despite of surgical treatment the rapid recurrentthyroid cancer either primary or metastatic had a poor prognosis withfatal outcome mainly in the presence liver cirrhosis and cardio-vascularco-morbidities.
Authors have no conflict of interests to disclose
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