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Neurol Med Chir (Tokyo) 45, 205¿208, 2005 Skull Metastasis From Thyroid Follicular Carcinoma With Difficult Diagnosis of the Primary Lesion —Case Report— Ismail AKDEMIR, Fatih S. EROL, Nusret AKPOLAT*, M. Faik OZVEREN, Murat AKFIRAT**, and Seyfettin YAHSI* Departments of Neurosurgery, *Pathology, and **Radiology, Firat University School of Medicine, Elazig, Turkey Abstract A 57-year-old male patient presented with an immobile ellipsoid mass of 6-cm diameter in the right occipitoparietal region. Cranial computed tomography showed the mass with dense contrast enhancement causing bone destruction. After embolization of the mass, total resection was performed. Histological examination showed the mass had a capsule, with no invasion of the dura mater or dermis, and the follicles of various sizes covered with mono-lined thyrocytes were full of colloid. Immunohistochemical examination showed positive staining for thyroglobulin. Postoperatively, levels of thyroid hormones were normal, and thyroid ultrasonography and technetium-99m scintigraphy showed no abnormalities. Fine needle aspiration biopsy performed at various locations of the thyroid gland revealed no atypical thyroid cells. Whole body technetium-99m scintigraphy found no abnormal bone involvement. The histological evidence was suggestive of follicular carcinoma metastasis. Surgical treatment was planned for the thyroid gland, but the patient did not consent. Two years later, the patient presented with the pain and sensitivity in the sacrum, the right iliac wing, and the right caput femoris. Computed tomography revealed lytic lesions in these areas. Bone metastases were identified. Whole body scintigraphy showed increased activity in these regions, but the cranium and all other tissues were normal. The patient underwent total thyroidectomy under a diagnosis of follicular carcinoma. The present case of a lytic skull lesion associated with normal thyroid tissue on admission but finally treated as follicular thyroid cancer emphasizes the difficulty in histological discrimination of follicular carcinoma from normal thyroid tissue. Key words: cranium, follicular carcinoma, metastasis Introduction Here we discuss a case of skull metastasis from thyroid follicular carcinoma which was difficult to identify. Skull metastases originating from tumors of the organs are rare. The most common forms are pulmonary and breast cancer metastases and prostate carcinomas.2,6) Bone metastasis of thyroid carcinoma is the second most common form of metastasis following pulmonary metastasis.1–3) However, metastases in the skull associated with carcinoma of the thyroid are rare, accounting for only 2.5% to 5.8% of cases. Solid masses causing lytic lesions in the cranium are suggestive of metastasis and present as various radiological findings with osteoblastic activity.4,6,7) The differential diagnosis of such lesions is essential for follow up and treatment. Received April 1, 2004; Accepted Case Report A 57-year-old male patient was referred to the Department of Neurosurgery, Firat University School of Medicine, with headache and a mass in the right occipitoparietal region. The mass had formed in the last year and rapidly grown within 2 months. Systemic and neurological examinations found no abnormalities except an immobile ellipsoid mass, 6 cm in diameter, in the occipitoparietal region. Computed tomography (CT) revealed a semisolid mass with contrast enhancement causing bone de- October 25, 2004 205 206 I. Akdemir et al. Fig. 1 Axial cranial computed tomography scan showing dense contrast enhancement of the mass causing bone destruction in the right occipitoparietal region. Fig. 2 Axial T2-weighted magnetic resonance image showing no invasion of the scalp and dura mater owing to the well-circumscribed capsule of the mass in the occipitoparietal region. struction in the right occipitoparietal region (Fig. 1). Magnetic resonance imaging showed absence of dura mater and scalp invasion of the richly vascular mass (Fig. 2). Digital subtraction angiography (DSA) showed the mass was supplied by the right occipital (75%), left occipital (20%), and right meningeal arteries (5%) (Fig. 3A), and drained through the Fig. 3 Preoperative digital subtraction angiograms showing the main blood supply through the occipital artery (A) and no venous connection of the mass with the superior sagittal sinus (B), and obstruction of the blood supply by embolization 3 days later (C). scalp veins, not through the sinuses (Fig. 3B). After DSA had confirmed embolization, the mass was totally resected (Figs. 3C and 4). The eroded bone Neurol Med Chir (Tokyo) 45, April, 2005 Thyroid Metastasis to the Skull Fig. 4 207 Photograph showing the mass after total removal, which was round, semi-solid, and 6 cm in diameter. parts were removed until normal bone structures were exposed. Histological examination showed the mass had a capsule, with no invasion of the dura mater or dermis, and the follicles of various sizes covered with mono-lined thyrocytes were full of colloid (Fig. 5B). Immunohistochemical staining showed diffuse positive staining for thyroglobulin (Fig. 5A). Postoperatively, his levels of thyroid hormones were normal. Thyroid ultrasonography and technetium-99m scintigraphy also showed no abnormalities. Fine needle aspiration cytology of various parts of thyroid gland revealed no atypical thyroid cells. Whole body technetium-99m scintigraphy showed no bone involvement. Hormone and physical examinations detected no abnormalities in the postoperative 3rd and 6th months. Physical examination of the cranium also found no abnormalities. Histological examination of the mass suggested follicular carcinoma metastasis, so surgical treatment of the thyroid gland was planned. However, the patient did not consent to the operation. Two years after the operation, the patient presented to our clinic with pain and sensitivity over the sacrum, the right iliac wing, and the right caput femoris. CT showed lytic lesions in these regions. Whole body scintigraphy showed increased activity in these regions, but the cranium treated before and other bone structures were normal. The patient underwent total thyroidectomy under a diagnosis of follicular carcinoma. Local radiotherapy was performed for the metastases. Follow-up examination after 1 year detected no recurrence. Neurol Med Chir (Tokyo) 45, April, 2005 Fig. 5 Photomicrographs revealing normal thyroid follicles of varying sizes filled with colloid, and positive immunohistochemical staining for thyroglobulin (A: ×200), and the cells well-restricted by the capsule, with no invasion of the dermis or the dura mater, and no malignant cytological features (B: HE stain, ×40). Discussion The largest series of skull metastases from thyroid carcinomas consists of 12 cases.3) The tumors caused only osteolytic lesions and formed soft but not painful hemispheric masses in the cranium. Most masses causing lytic lesions in various locations are follicular carcinomas, which are vascular tumors with strong arterial pulsations, and regular and solid margins.1,3) Generally, the margins of metastatic masses are irregular. Multiple bone destruction and secondary thickness occur. In our case, the tumor was a solid, regular, and vascular formation which caused an extensive bone defect in the parietooccipital region. The mean age of the patients is 60 years. The incidence is higher in females than in males. The incidence of metastases, 208 I. Akdemir et al. other than thyroid metastases, significantly increases over 40 years of age with an equal ratio of males and females.7) Metastatic tumors with unidentified primary tumor histology have been reported in patients with normal thyroid glands.1,3) Follicular carcinoma cannot be discriminated from normal thyroid tissue based on morphological criteria, so the diagnosis of carcinoma can be established only in the presence of a capsule or vessel invasion. Fine needle aspiration cytology can sample the cells but not the capsule or the vessels. Therefore, thyroid tumor tissue containing follicular cells is difficult to identify because of the normal appearance. The changes in the thyroid gland and hormone profile or histological studies of cross-sections obtained at thyroidectomy may help to identify the tumor. Increased scintigraphic activity is important evidence for both thyroid tissue carcinomas and metastases. Distant organ lesions characterized as ectopic thyroid tissue include cases with either normal tissue or carcinoma.5) Investigation of follicular carcinoma at embryologically impossible locations for ectopic thyroid tissue may provide more specific findings. Palpable lytic tumor of the scalp is the principal physical manifestation, whereas cranial nerve dysfunction, focal brain symptoms, or symptoms due to increased intracranial pressure are rare. However, the most important problem is the bone defects requiring extensive bone resection. The mean period until the diagnosis of thyroid tumors in patients undergoing thyroidectomy is 14.5 years, and until detection of the skull metastasis is 23.3 years.3) However, the mean period until diagnosis of the definite metastatic focus ranges from 5 months to 4 years.3) In our case, the period until diagnosis of the definite metastatic focus was 2 years. Despite slow progression, the masses can adopt a variable and insidious clinical profile. In this period, the clinical profile may be masked a little longer since no changes may take place in the thyroid gland and hormone. Strikingly, in our case, the metastasis involved a different bone focus within 1 year, although there was no recurrence in the first focus. The optimum therapy for patients with bone metastasis due to thyroid tissue tumors involves complete excision of the thyroid gland together with all the probable metastatic foci and regional lymph nodes, and application of local radiotherapy.3) Frequent follow-up examination is also recommended. The primary focus of thyroid metastasis, which causes large bone defects, is difficult to define.2,5) Various reports describe 3 or 4 years of treatment and follow up for these cases.2,3) These tumors are rare, difficult to define histologically, and may often mimic other bone pathologies, so critical clinical diagnosis and aggressive therapy are required. References 1) 2) 3) 4) 5) 6) 7) Inci S, Akbay A, Bertan V, Gedikoglu G, Onol B: Solitary skull metastasis from occult thyroid carcinoma. J Neurosurg Sci 38: 63–66, 1994 McCormack KR: Bone metastases from thyroid carcinoma. Cancer 19: 181–184, 1966 Nagamine Y, Suzuki J, Katakura R, Yoshimoto T, Matoba N, Takaya K: Skull metastasis of thyroid carcinoma study of 12 cases. J Neurosurg 63: 526–531, 1985 Osborn AG: Brain tumors and tumorlike processes, in: Diagnostic Neuroradiology. St Louis, Mosby, 1994, pp 516–517 Ruchti C, Balli-Antunes M, Gerber HA: Follicular tumor in the sellar region without primary cancer of the thyroid. Heterotopic carcinoma? Am J Clin Pathol 87: 776–780, 1987 Turner O, German WJ: Metastases in the skull from carcinoma of the thyroid. Surgery 9: 403–414, 1941 Wilkins RH, Rengachary SS: Tumors of the skull, in: Neurosurgery, ed 2. New York, McGraw-Hill, 1996, pp 1503–1529 Address reprint requests to: I. Akdemir, M.D., Neurosurgery Department, Arastirma Hospital, Firat University School of Medicine, 23100 Elazig, Turkey. e-mail: akdemir23@yahoo.com Neurol Med Chir (Tokyo) 45, April, 2005