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      A ‘silent’ skull metastatic follicular thyroid carcinoma mimicking as a benign scalp tumor in a pregnant woman

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          Summary

          Thyroid cancer with cranial metastasis in a pregnant woman is very rare. In the literature, most cases are diagnosed early from neurogenic signs or symptomatic thyroid gland. Pregnancy also contributes to a hesitation toward early surgical and medical treatments. We reported a scalp tumor in a physically healthy 37-year-old pregnant female with a follicular thyroid carcinoma (FTC) with lung, bone and cranial metastasis in initial presentation. Silent neurogenic and physical examinations make an early diagnosis very challenging. Resection of scalp and intracranial tumor, a thyroidectomy, post-operative radioactive iodine therapy and tyrosine kinase inhibitors were employed as treatment. The scalp tumor was confirmed as a metastatic follicular thyroid carcinoma via positive immunoreactivity for thyroglobulin and thyroid transcription factor 1 in tumor cells. Blood examination revealed an elevated thyroglobulin level (>5335 ng/mL). The patient was discharged without any neurological deficit. An asymptomatic scalp tumor in a pregnant woman with a normal thyroid disease history needs differential diagnosis from intracranial origin. Rapid progression and an elevated thyroglobulin level are the indicators that further image study is needed. Aggressive surgical excision of resectable thyroid gland and metastatic tumor are essential for a longer survival rate. There is nothing to indicate that a post-partum operation will worsen prognosis.

          Learning points:
          • Follicular thyroid cancer with cranial metastasis in initial presentation can be asymptomatic.

          • Follicular thyroid cancer with cranial metastasis in a pregnant woman can be treated after delivery.

          • Rapid enlargement of scalp tumor is indicated for further image study even in a patient without any neurological deficit.

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          Most cited references14

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          Treatment with tyrosine kinase inhibitors for patients with differentiated thyroid cancer: the M. D. Anderson experience.

          Until recently, treatment options for patients with progressive, radioactive iodine-resistant differentiated thyroid cancer (DTC) have been limited. In our clinical practice, we have begun to use sorafenib and sunitinib for patients with progressive DTC who are not able or willing to participate in clinical trials. In this paper, we describe the University of Texas M. D. Anderson Cancer Center's experience with the off-label use of these tyrosine kinase inhibitors for DTC. Adult patients were included if they had a diagnosis of radioactive iodine-refractory DTC, were treated with single agent sorafenib or sunitinib, and had both baseline and at least one follow-up scan for restaging purposes. All imaging data were collected, as well as the TSH-suppressed thyroglobulin (Tg) levels corresponding to each scan date. The primary endpoints were radiographic response and progression-free survival (PFS). Secondary objectives were tissue-specific radiographic responses and correlation of Tg with overall response. We identified 33 patients from our clinical database. Fifteen patients (nine women, six men) met inclusion criteria, with a median age of 61 yr (range, 38-83 yr). Eight patients had papillary and seven had follicular thyroid carcinoma. Sorafenib was used in 13 and sunitinib in two, including one patient who failed prior sorafenib therapy. All patients had evidence of progressive disease (PD) before start of therapy, with a median PFS of only 4 months. Best response in target lesions was: partial response (PR) in three (20%), stable disease (SD) in nine (60%), and PD in three (20%). Clinical benefit (PR+SD) was 80%. The sunitinib patient previously refractory to sorafenib had a 38% reduction in tumor size. The most noticeable organ-specific response was observed in lung (median change, -22%) compared to lymph nodes (median change, 0%). Pleural disease and nonirradiated bone metastases demonstrated PD. All histological subtypes had similar responses. The median PFS was 19 months. The median overall survival has not yet been reached, but at 2 yr of follow-up, overall survival is 67%. Log Tg correlated with radiographic response (P = 0.0005). Sorafenib and sunitinib appear to be effective in patients with widely metastatic, progressive DTC, with most patients achieving SD or PR, despite having PD at baseline. The most noticeable responses occurred in the lungs in contrast with minimal changes in nodal metastases and PD in pleural and nonirradiated bone metastases, suggesting a tissue-specific response to therapy. Log Tg significantly correlated with response to treatment and therefore may have value as a surrogate marker of response.
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            Management of brain metastases from thyroid carcinoma: a study of 16 pathologically confirmed cases over 25 years.

            Brain metastasis is an uncommon, morbid complication of metastatic thyroid carcinoma. Because of its rarity, management often is problematic. To help contribute to the management of this disease entity, the authors present herein what to their knowledge is the largest series reported to date in which all patients had biopsy proven confirmation of their brain metastases. The authors report a series of 16 patients with metastatic thyroid carcinoma to the brain who were treated between 1976-2000. The Mayo Clinic database was used to locate and review charts and radiology and pathology reports, and all biopsy specimens were reviewed by one pathologist. The histologic types of carcinoma included 10 papillary carcinomas, 2 follicular carcinomas, 1 Hürthle cell carcinoma, 1 medullary carcinoma, 1 insular carcinoma, and 1 anaplastic carcinoma. Given the small sample size, statistical analyses were not performed. Surgical resection of brain metastases was associated with a trend toward longer survival (20.8 months vs. 2.7 months for no surgical intervention in selected patients) Whole brain external beam radiation therapy produced disease regression in three of the four evaluable patients. Gamma knife radiosurgery and radioactive iodine therapy appear to play limited, but beneficial, therapeutic roles. Overall, survival after the diagnosis of brain metastasis is reported to be longer than that noted with other solid tumors (17.4 months), and the majority of patients die of their extracranial disease (85% in the current series). The results of the current study indicate that local therapies appear to control brain metastases in the large majority of thyroid carcinoma patients with metastases to the brain. Copyright 2003 American Cancer Society.
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              Differentiated thyroid carcinoma presenting with distant metastasis at initial diagnosis clinical outcomes and prognostic factors.

              Distant metastasis (DM) is seldom observed at initial presentation of differentiated thyroid carcinoma (DTC), making it difficult to assess the clinical characteristics and treatment outcomes of DTC patients with DM. We therefore retrospectively assessed these parameters in DTC patients who presented with DM between July 1994 and December 2007. In addition, we compared biologic behaviors and prognostic factors between patients presenting with DM and those developing DM after initial treatment. Among 1560 DTC patients who underwent thyroidectomies during the 13.5-year study period, 91 patients were included in this study; 52 patients (3.4%) displayed DM at initial presentation and 39 (2.5%) developed DM after initial 131 radiacitive iodine (RI) treatment. Metastatic lesions were treated with high dose RI (94.5%), surgical resection (14.3%), external beam radiation therapy (31.9%), embolization of feeding vessels (1.1%), and/or chemotherapy (1.1%). Median duration of follow-up was 75 months (range, 12-158 months). Mean patient age was 57 years (range, 13-80 years), and the female-to-male ratio was 2.03:1. Metastases were detected in the lung only (68.1%), bone only (16.5%), and multiple sites (15.4%). When clinical and tumor characteristics were considered, metastatic lesion iodine avidity was significantly higher in patients presenting with DM. At 5 and 10 years, the overall survival (OS) rates in patients presenting with DM were 83.8% and 72.1%, respectively, and the disease-specific survival (DSS) rates were 68.5% and 26.8%. OS did not differ significantly between patients presenting with DM and those developing DM after initial treatment. However, those with initial DM enjoyed significantly improved DSS compared with patients who developed DM after initial treatment. At last follow-up, 22 patients (24.2%) were alive without disease, 48 (52.7%) were alive with disease, and 21 (23.1%) had died of disease in study patients. Multivariate Cox's regression analyses showed that complete local control was a significant predictor of OS and DSS in all study patients. In patients developing DM after initial treatment, metastatic lesion iodine avidity was also associated with both OS and DSS. In contrast, avidity was associated only with DSS in patients presenting with DM. Our study showed that DTC patients presenting with initial DM appear to have relatively favorable outcomes compared with DTC patients who developed DM after initial treatment. Complete local control may be the most important prognostic indicator in all DM patients. Metastatic lesion iodine avidity had a significant impact on both OS and DSS in patients developing DM after initial treatment, but significantly influenced only DSS in patients presenting with initial DM.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                12 January 2017
                2017
                : 2017
                : 16-0100
                Affiliations
                [1 ]Departments of Plastic and Reconstructive Surgery
                [2 ]Departments of Neurosurgery
                [3 ]Departments of Pathology , China Medical University Hospital, China Medical University, Taichung, Taiwan
                Author notes
                Correspondence should be addressed to H-H Chen; Email: scapulachenhh@ 123456yahoo.com.tw
                Article
                EDM160100
                10.1530/EDM-16-0100
                5292982
                28203373
                af8cb856-3853-4297-ab29-10b4f76c73b6
                This is an Open Access article distributed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License.

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License .

                History
                : 23 October 2016
                : 28 November 2016
                Categories
                Unique/Unexpected Symptoms or Presentations of a Disease

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