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Tumor Within Another Tumor: An Unusual Case of Metastatic Poorly Differentiated Thyroid Carcinoma Within an Intracranial Meningioma Min Ling, Tang Wong, Chuong D. Bui, Seymour Atlas, Jing J. Li, Tao Yang, Sandra Harvey, and Jeff R. Flack Department of Diabetes and Endocrinology (M.L., T.W., J.R.F.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia; University of New South Wales (M.L., T.W., S.A., J.R.F.), Sydney, New South Wales 2052, Australia; Department of Nuclear Medicine and Ultrasound (C.D.B.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia; Department of Radiology (S.A.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia; Department of Pathology (J.J.L., T.Y.), Liverpool Hospital, Sydney, New South Wales 2170, Australia; and Department of Oncology (S.H.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia
60-year-old woman with progressive iodine-refractory thyroid cancer with skeletal and pulmonary metastases developed right-sided ataxia. Multifocal papillary and poorly differentiated thyroid carcinoma (Figure 1, A and B) were diagnosed 6 years earlier at total thyroidectomy for multinodular goiter. Wholebody scintigraphy following course six of I131 therapy (cumulative dose, 29.7 GBq) identified a new iodineavid posterior fossa lesion (Figure 2A) with serum thyroglobulin levels of 1224 ng/mL (unstimulated) and 1334 ng/mL (stimulated). Prednisolone at a daily dose of 50 mg was tapered after treatment over a duration of 2 weeks for known metastatic vertebral disease. Magnetic resonance imaging of the brain showed a posterior fossa dural-based mass (20 ⫻ 23 ⫻ 25 mm) consistent with a meningioma, apart from the uncommon finding of heterogeneous enhancement (Figure 2B). With provisional diagnosis of metastatic disease in posterior fossa, prednisolone was changed to dexamethasone 4 mg twice daily and maintained until her metastasectomy 1 month later. Histopathology confirmed World Health Organization grade-1 meningioma (Figure 1C) with positive epithelial membrane antigen staining (data not shown) and containing a metastatic poorly differentiated thyroid carcinoma (Figure 1D). Immunohistochemistry stained positive for thyroid
A
transcription factor-1, thyroglobulin (Figure 1E), and cytokeratin-MNF116 (Figure 1F). Despite symptomatic response from metastasectomy and external beam radiotherapy, she died 12 months later from complications of cauda equina syndrome. Undiagnosed thyroid cancer can present as an intracranial metastasis mimicking meningioma (1). Furthermore, meningioma is the most common intracranial tumor to harbor metastasis, often of lung and breast origin (2). Although the mechanism behind tumor-totumor metastasis is unknown, thyroid cancer and meningioma share biological, metabolic, molecular, and hormonal factors, including the presence of psammoma bodies and somatostatin receptors on both tumors, thyroid hormone receptors on meningioma cells, and possible linked gene mutations (3). Meningiomas themselves can exhibit iodine avidity on scintigraphy, in the absence of thyroid cancer due to edema and high vascularity (4, 5). This is the first reported case of poorly differentiated thyroid cancer metastasizing within an intracranial meningioma from initial multifocal mixed thyroid cancer. Although meningiomas may physiologically concentrate I131, our case highlights the importance of excluding metastatic disease in similar patients with apparently benign lesions (5).
ISSN Print 0021-972X ISSN Online 1945-7197 Printed in U.S.A. Copyright © 2014 by the Endocrine Society Received February 2, 2014. Accepted June 18, 2014. First Published Online June 30, 2014
doi: 10.1210/jc.2014-1310
J Clin Endocrinol Metab, October 2014, 99(10):3513–3514
jcem.endojournals.org
The Endocrine Society. Downloaded from press.endocrine.org by [${individualUser.displayName}] on 14 January 2015. at 04:05 For personal use only. No other uses without permission. . All rights reserved.
3513
3514
Ling et al
Thyroid Cancer Metastasis within Meningioma
J Clin Endocrinol Metab, October 2014, 99(10):3513–3514
Figure 1. Histopathology of primary thyroid carcinoma (A and B), histopathology/immunohistochemistry of intracranial lesion (C–E), and interface of two tumors (F). A, Primary thyroid carcinoma containing poorly differentiated component with insular pattern (arrows) and necrosis (arrowheads). B, Papillary carcinoma component with typical nuclear features: overlapping/ground-glass nuclei and nuclear grooves (arrows). C, Meningioma showing mixed meningothelial and fibrous patterns, with focal calcification; confirmed also by positive epithelial membrane antigen staining for the meningioma cells (data not shown). D, Metastatic thyroid carcinoma (arrow) in meningioma tissue (arrowhead). E, Metastatic thyroid carcinoma positive for thyroid transcription factor-1 (inset, also positive for thyroglobulin). F, Interface of tumors: cytokeratin positive in thyroid carcinoma (left), but negative in meningioma (right).
Acknowledgments Address all correspondence and requests for reprints to: Tang Wong, Department of Diabetes and Endocrinology, BankstownLidcombe Hospital, Eldridge Road, Bankstown, New South Wales 2200, Australia. E-mail: [email protected].
No grants or fellowships supported the writing of the paper. Disclosure Summary: The authors have nothing to declare.
Figure 2. A, Post-therapy (course six of I131, 5.53 GBq) whole-body I131 scintigraphy demonstrating a new intensely iodine-avid intracranial lesion (arrow), along with known lung, mediastinal, and bone lesions. B, Post-contrast T1-weighted coronal magnetic resonance imaging of the brain showing a right dural-based posterior fossa mass with heterogeneous enhancement (arrowhead).
References 1. Portocarrero-Ortiz L, Garcia-Lopez R, Romero-Vargas S, et al. Thyroid follicular carcinoma presenting as skull and dural metastasis mimicking a meningioma: a case report. J Neurooncol. 2009;95:281–284. 2. Moody P, Murtagh K, Piduru S, Brem S, Murtagh R, Rojiani AM. Tumor-to-tumor metastasis: pathology and neuroimaging considerations. Int J Clin Exp Pathol. 2012;5(4):367–373. 3. Sughrue ME, Kane AJ, Shangari G, Parsa AT, Berger MS, McDermott MW. Prevalence of previous extracranial malignancies in a series of 1228 patients presenting with meningioma. J Neurosurg. 2010; 113(5):1115–1121. 4. Preisman RA, Halpern SE, Shishido R, Waltz T, Callipari F, Reit R. Uptake of 131I by a papillary meningioma. AJR Am J Roentgenol. 1977;129(2):349 –350. 5. Sinha P, Conrad GR, Holzhauer M. Incidental detection of a falx meningioma on posttherapy radioiodide whole-body imaging. Clin Nucl Med. 2002;27(12):916–917.
The Endocrine Society. Downloaded from press.endocrine.org by [${individualUser.displayName}] on 14 January 2015. at 04:05 For personal use only. No other uses without permission. . All rights reserved.
i n
F E A T U R E
E n d o c r i n o l o g y
Tumor Within Another Tumor: An Unusual Case of Metastatic Poorly Differentiated Thyroid Carcinoma Within an Intracranial Meningioma Min Ling, Tang Wong, Chuong D. Bui, Seymour Atlas, Jing J. Li, Tao Yang, Sandra Harvey, and Jeff R. Flack Department of Diabetes and Endocrinology (M.L., T.W., J.R.F.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia; University of New South Wales (M.L., T.W., S.A., J.R.F.), Sydney, New South Wales 2052, Australia; Department of Nuclear Medicine and Ultrasound (C.D.B.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia; Department of Radiology (S.A.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia; Department of Pathology (J.J.L., T.Y.), Liverpool Hospital, Sydney, New South Wales 2170, Australia; and Department of Oncology (S.H.), Bankstown-Lidcombe Hospital, Sydney, New South Wales 2200, Australia
60-year-old woman with progressive iodine-refractory thyroid cancer with skeletal and pulmonary metastases developed right-sided ataxia. Multifocal papillary and poorly differentiated thyroid carcinoma (Figure 1, A and B) were diagnosed 6 years earlier at total thyroidectomy for multinodular goiter. Wholebody scintigraphy following course six of I131 therapy (cumulative dose, 29.7 GBq) identified a new iodineavid posterior fossa lesion (Figure 2A) with serum thyroglobulin levels of 1224 ng/mL (unstimulated) and 1334 ng/mL (stimulated). Prednisolone at a daily dose of 50 mg was tapered after treatment over a duration of 2 weeks for known metastatic vertebral disease. Magnetic resonance imaging of the brain showed a posterior fossa dural-based mass (20 ⫻ 23 ⫻ 25 mm) consistent with a meningioma, apart from the uncommon finding of heterogeneous enhancement (Figure 2B). With provisional diagnosis of metastatic disease in posterior fossa, prednisolone was changed to dexamethasone 4 mg twice daily and maintained until her metastasectomy 1 month later. Histopathology confirmed World Health Organization grade-1 meningioma (Figure 1C) with positive epithelial membrane antigen staining (data not shown) and containing a metastatic poorly differentiated thyroid carcinoma (Figure 1D). Immunohistochemistry stained positive for thyroid
A
transcription factor-1, thyroglobulin (Figure 1E), and cytokeratin-MNF116 (Figure 1F). Despite symptomatic response from metastasectomy and external beam radiotherapy, she died 12 months later from complications of cauda equina syndrome. Undiagnosed thyroid cancer can present as an intracranial metastasis mimicking meningioma (1). Furthermore, meningioma is the most common intracranial tumor to harbor metastasis, often of lung and breast origin (2). Although the mechanism behind tumor-totumor metastasis is unknown, thyroid cancer and meningioma share biological, metabolic, molecular, and hormonal factors, including the presence of psammoma bodies and somatostatin receptors on both tumors, thyroid hormone receptors on meningioma cells, and possible linked gene mutations (3). Meningiomas themselves can exhibit iodine avidity on scintigraphy, in the absence of thyroid cancer due to edema and high vascularity (4, 5). This is the first reported case of poorly differentiated thyroid cancer metastasizing within an intracranial meningioma from initial multifocal mixed thyroid cancer. Although meningiomas may physiologically concentrate I131, our case highlights the importance of excluding metastatic disease in similar patients with apparently benign lesions (5).
ISSN Print 0021-972X ISSN Online 1945-7197 Printed in U.S.A. Copyright © 2014 by the Endocrine Society Received February 2, 2014. Accepted June 18, 2014. First Published Online June 30, 2014
doi: 10.1210/jc.2014-1310
J Clin Endocrinol Metab, October 2014, 99(10):3513–3514
jcem.endojournals.org
The Endocrine Society. Downloaded from press.endocrine.org by [${individualUser.displayName}] on 14 January 2015. at 04:05 For personal use only. No other uses without permission. . All rights reserved.
3513
3514
Ling et al
Thyroid Cancer Metastasis within Meningioma
J Clin Endocrinol Metab, October 2014, 99(10):3513–3514
Figure 1. Histopathology of primary thyroid carcinoma (A and B), histopathology/immunohistochemistry of intracranial lesion (C–E), and interface of two tumors (F). A, Primary thyroid carcinoma containing poorly differentiated component with insular pattern (arrows) and necrosis (arrowheads). B, Papillary carcinoma component with typical nuclear features: overlapping/ground-glass nuclei and nuclear grooves (arrows). C, Meningioma showing mixed meningothelial and fibrous patterns, with focal calcification; confirmed also by positive epithelial membrane antigen staining for the meningioma cells (data not shown). D, Metastatic thyroid carcinoma (arrow) in meningioma tissue (arrowhead). E, Metastatic thyroid carcinoma positive for thyroid transcription factor-1 (inset, also positive for thyroglobulin). F, Interface of tumors: cytokeratin positive in thyroid carcinoma (left), but negative in meningioma (right).
Acknowledgments Address all correspondence and requests for reprints to: Tang Wong, Department of Diabetes and Endocrinology, BankstownLidcombe Hospital, Eldridge Road, Bankstown, New South Wales 2200, Australia. E-mail: [email protected].
No grants or fellowships supported the writing of the paper. Disclosure Summary: The authors have nothing to declare.
Figure 2. A, Post-therapy (course six of I131, 5.53 GBq) whole-body I131 scintigraphy demonstrating a new intensely iodine-avid intracranial lesion (arrow), along with known lung, mediastinal, and bone lesions. B, Post-contrast T1-weighted coronal magnetic resonance imaging of the brain showing a right dural-based posterior fossa mass with heterogeneous enhancement (arrowhead).
References 1. Portocarrero-Ortiz L, Garcia-Lopez R, Romero-Vargas S, et al. Thyroid follicular carcinoma presenting as skull and dural metastasis mimicking a meningioma: a case report. J Neurooncol. 2009;95:281–284. 2. Moody P, Murtagh K, Piduru S, Brem S, Murtagh R, Rojiani AM. Tumor-to-tumor metastasis: pathology and neuroimaging considerations. Int J Clin Exp Pathol. 2012;5(4):367–373. 3. Sughrue ME, Kane AJ, Shangari G, Parsa AT, Berger MS, McDermott MW. Prevalence of previous extracranial malignancies in a series of 1228 patients presenting with meningioma. J Neurosurg. 2010; 113(5):1115–1121. 4. Preisman RA, Halpern SE, Shishido R, Waltz T, Callipari F, Reit R. Uptake of 131I by a papillary meningioma. AJR Am J Roentgenol. 1977;129(2):349 –350. 5. Sinha P, Conrad GR, Holzhauer M. Incidental detection of a falx meningioma on posttherapy radioiodide whole-body imaging. Clin Nucl Med. 2002;27(12):916–917.
The Endocrine Society. Downloaded from press.endocrine.org by [${individualUser.displayName}] on 14 January 2015. at 04:05 For personal use only. No other uses without permission. . All rights reserved.
