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      Late bone metastasis from an apparently benign oncocytic follicular thyroid tumor

      case-report

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          Abstract

          A man underwent total thyroidectomy for goiter when he was 62 years old. The pathology report informed on a 5.5 cm oncocytic follicular adenoma and a 3.5 mm papillary microcarcinoma. Due to the papillary tumor, he was treated with ablative radioiodine therapy and suppressive doses of levothyroxine. After uneventful follow-up for 9 years, increased levels of serum thyroglobulin were detected. Further imaging studies including a whole body scan (WBS) after an empirical dose of 200 mCi 131I were negative. Two years later, a 99mTc SestaMIBI WBS and a 2-[ 18F]-fluoro-2-deoxy- d-glucose positron-emission tomography showed a well-delimited focal uptake in the right femur. A bone biopsy of the lesion demonstrated metastasis of follicular thyroid carcinoma. Retrospective histological reexamination of available material from the primary oncocytic thyroid tumor failed to reveal definitive traits of malignancy.

          Learning points

          • Oncocytic follicular thyroid tumors are a relatively uncommon variant of follicular thyroid neoplasms mostly composed of distinctive large oxyphilic cells (Hürthle cells).

          • Criteria for the distinction between benign and malignant oncocytic neoplasms are not different from those used in the diagnosis of ordinary follicular tumors.

          • Some cases of apparently benign oncocytic neoplasms have been found to develop malignant behavior.

          • Search to rule out vascular and capsular invasion should be particularly exhaustive in histological assessment of oncocytic thyroid tumors.

          • Even so, long-term surveillance remains appropriate for patients with large apparently benign oncocytic tumors.

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          Most cited references7

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          Prognostic factors of recurrence in encapsulated Hurthle cell carcinoma of the thyroid gland: a clinicopathologic study of 50 cases.

          Follicular carcinomas of the thyroid gland, including its oncocytic variant (so-called Hurthle cell carcinoma), are subdivided into the indolent encapsulated ("minimally invasive") and the clinically aggressive widely invasive tumors. There are, however, cases of encapsulated follicular carcinoma that recur and metastasize. Identifying these cases at the time of diagnosis is crucial for prognostic and therapeutic considerations. Because to the authors' knowledge most studies do not focus exclusively on the encapsulated Hurthle cell carcinoma (EHC), the current study attempted to identify predictors of recurrence in EHC. A tumor was defined as EHC if it was encapsulated, macroscopically well defined with microscopic but no macroscopic evidence of vascular or capsular invasion, and composed of > 75% follicular oncocytic cells. Retrospective chart review and microscopic examination identified 50 primary tumors meeting the above criteria at the Memorial Sloan-Kettering Cancer Center between 1967 and 2005. The cases were analyzed for various histologic and clinical parameters. Each parameter was correlated with recurrence-free survival (RFS). Seven of 50 (14%) patients developed disease recurrence. All patients who developed recurrence were found to have a high number of foci of vascular invasion (> or = 4). In univariate analysis, > or = 4 foci of vascular invasion (P 4 cm (P = .049), the presence of mitosis (P = .018), and a solid/trabecular growth pattern (P = .009) were found to be correlated with a decreased RFS. Extensive capsular invasion, gender, and age did not confer a statistically higher recurrence rate. The finding of a solid/trabecular growth and mitosis correlated with the presence of numerous foci (> or = 4) of vascular invasion (P = .01 and P = .005, respectively). A diligent search for vascular invasion is recommended in EHC that display mitosis or a solid/trabecular growth pattern. The presence of > or = 4 foci of vascular invasion should alert the pathologist and the clinician to a significantly higher risk of recurrence in EHC. 2006 American Cancer Society
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            Diagnostic accuracy and prognostic value of 18F-FDG PET in Hürthle cell thyroid cancer patients.

            Hürthle cell carcinoma is an uncommon and occasionally aggressive differentiated thyroid cancer associated with increased mortality compared with other differentiated thyroid malignancies. Because it generally has lower iodine avidity, 18F-FDG PET has been suggested as a more accurate imaging modality. However, there is limited information with regard to the true diagnostic accuracy and prognostic value of 18F-FDG PET in this disease. All patients with Hürthle cell thyroid cancer who underwent their first 18F-FDG PET scan between May 1996 and February 2003 were identified retrospectively. 18F-FDG PET scans were reviewed and compared with all available imaging studies, including CT, ultrasound, and radioiodine scintigraphy (RIS). Abnormal 18F-FDG uptake was assessed visually and by measuring the maximum standardized uptake value (SUVmax) of the most intense lesion. Clinical follow-up for at least 1 y or until death was required for inclusion. Forty-four patients met inclusion criteria. The median follow-up was 2.9 y. There were 24 positive and 20 negative 18F-FDG PET scans with 1 false-positive and 1 false-negative study, resulting in a diagnostic sensitivity of 95.8% and a specificity of 95%. In 5 of 11 patients who had both positive CT and 18F-FDG PET findings, 18F-FDG PET revealed additional sites of disease. Furthermore, 18F-FDG PET correctly classified as negative 3 patients with false-positive CT findings. In 3 of 6 patients with positive RIS, 18F-FDG PET revealed additional sites of metastatic disease. Ten patients with positive 18F-FDG PET had negative RIS. Only 1 patient with negative 18F-FDG PET had positive RIS. The SUVmax also provided prognostic information: In a stepwise fashion, each increase in intensity by SUVmax unit was associated with a 6% increase in mortality (P 10 (P < 0.01). 18F-FDG PET has excellent diagnostic accuracy in Hürthle cell thyroid cancer patients, improving on CT and RIS. Intense 18F-FDG uptake in lesions is an indicator of a poor prognosis. Our data suggest that all patients with Hürthle cell thyroid cancer should undergo 18F-FDG PET as part of their initial postoperative staging and periodically to screen for occult recurrence, particularly in patients with elevated serum thyroglobulin.
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              Older age and larger tumor size predict malignancy in hürthle cell neoplasms of the thyroid.

              Hürthle cell neoplasms (HCNs) are rare tumors of the thyroid gland. The definitive treatment for Hürthle cell carcinoma (HCC) is total thyroidectomy, while thyroid lobectomy is adequate for Hürthle cell adenoma (HCA). However, differentiating HCC from HCA either before or during surgery is a challenge. The purpose of this study was to identify factors that predict malignancy in patients with HCN. Between May 1994 and January 2007, 1,199 patients underwent thyroid surgery at an academic medical center. Medical records of 55 consecutive patients who underwent thyroid resections for the preoperative diagnosis of HCN were reviewed. Of the 55 patients with HCN, 46 (84%) had adenomas and 9 (16%) had carcinomas. Patients with HCC were significantly older than those with HCA (66 +/- 6 years versus 53 +/- 2 years, P = 0.01). Patients with carcinoma also had significantly larger thyroid nodules (4.5 +/- 0.7 cm versus 2.5 +/- 0.2 cm, P < 0.001). All HCNs less than 2 cm in diameter were benign. The malignancy rate increased with nodule size: 18% of nodules measuring 2-4 cm, and 44% of those larger than 4 cm were HCC. One patient with HCC had recurrence of the disease, but there were no disease-related deaths. Advanced patient age and larger nodule size are two important factors that predict malignancy in patients with HCN. In patients with these and other known risk factors for HCC, total thyroidectomy should be considered.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                edm
                EDM Case Reports
                Endocrinology, Diabetes & Metabolism Case Reports
                BioScientifica (Bristol )
                2052-0573
                30 September 2013
                2013
                : 2013
                : 130051
                Affiliations
                [1]Section of Endocrinology and Nutrition Complejo Hospitalario Universitario Insular Materno-Infantil Avenida Marítima del Sur, s/n. 35016, Las Palmas de Gran CanariaSpain
                [1 ]Service of Pathology Complejo Hospitalario Universitario Insular Materno-Infantil Avenida Marítima del Sur, s/n. 35016, Las Palmas de Gran CanariaSpain
                [2 ]Service of Nuclear Medicine Hospital General de Gran Canaria Dr Negrín. Plaza Barranco de la Ballena s/n. 35012, Las Palmas de Gran CanariaSpain
                [3 ]Service of Nuclear Medicine Complejo Hospitalario Universitario Insular Materno-Infantil Avenida Marítima del Sur, s/n. 35016, Las Palmas de Gran CanariaSpain
                Author notes
                Correspondence should be addressed to M Boronat Email: mborcor@ 123456ono.com
                Article
                EDM130051
                10.1530/EDM-13-0051
                3922002
                24616777
                e2150331-1311-41a2-a4b8-fc07a456e290
                © 2013 The authors

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

                History
                : 14 September 2013
                : 17 September 2013
                Categories
                Error in Diagnosis/Pitfalls and Caveats

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