□
CASE REPORT
□
Choroid Plexus Metastasis of Follicular Thyroid Carcinoma Diagnosed due to Intraventricular Hemorrhage Toru Umehara 1, Yoshiko Okita 1, Masahiro Nonaka 1, Kosuke Mori 1, Yonehiro Kanemura 1, Yoshinori Kodama 2, Masayuki Mano 2, Ikuo Kudawara 3 and Shin Nakajima 1
Abstract Choroid plexus metastasis (CPM) is extremely rare and originates most frequently from renal cell carcinoma (RCC). We herein report the case of a 58-year-old man who developed a solitary CPM lesion derived from follicular thyroid carcinoma in addition to intraventricular hemorrhage. Computed tomography revealed acute hydrocephalus as a result of the hemorrhage, and we planned endoscopic hematoma evacuation. Since it was too difficult to reach the hematoma, we considered the possibility of a neoplasm and performed a biopsy of the lesion, the results of which led to an accurate diagnosis of CPM in this case. We also review previous reports of CPM originating from thyroid carcinoma compared with RCC. Key words: intraventricular hemorrhage, choroid plexus metastasis, follicular thyroid carcinoma (Intern Med 54: 1297-1302, 2015) (DOI: 10.2169/internalmedicine.54.3560)
we describe the sixth case of FTC metastasizing to the choroid plexus in a patient presenting with intraventricular hemorrhage (IVH) 13 years after total thyroidectomy.
Introduction The incidence of thyroid carcinoma is fairly low, accounting for only approximately 1% of new cases of malignant disease worldwide. The majority (94%) of such cases involve differentiated thyroid carcinoma (DTC), either papillary thyroid carcinoma or follicular thyroid carcinoma (FTC) (1), which is generally associated with an indolent disease course. Indeed, the prognosis of DTC remains excellent, with the 10-year cancer-specific survival rate reaching 90% (2, 3). Local invasion is the usual mode of spread, followed by dissemination via the lymphatic or hematogenous route. The lungs and bone are the most common sites of distant metastasis. Conversely, brain metastasis of DTC is very rare, occurring in approximately 0.9% of the patient population, and is a marker of a poor prognosis (4). The disease-specific mortality rate of DTC metastasis is 67%, with a median product-limit survival of 12.4 months (4-7). In addition, among types of brain metastasis cases, choroid plexus metastasis (CPM) is extremely rare, with only five cases reported in the literature to date (8-12). In this report,
Case Report We herein report the case of a 58-year-old man who presented with severe headache, vomiting and somnolence. He had undergone total thyroidectomy for FTC 13 years prior to presentation and had received radiation therapy for multiple bone metastases in the right clavicle, right fifth rib and lumbar vertebrae (L2-3). A clinical examination performed on admission revealed a Glasgow coma scale score of 14 points (E4, V4, M6), with no focal neurological deficits. A computed tomography (CT) scan of the head showed IVH in the third ventricle without hydrocephalus (Fig. 1), whereas subsequent three-dimensional CT angiography (CTA) revealed no evidence of intracranial vascular abnormalities, initially suspected to be the source of bleeding. Half a day after presentation, the patient exhibited slightly disturbed consciousness resulting from hydrocephalus. Accordingly, we planned endoscopic hematoma evacuation for
１
Department of Neurosurgery, National Hospital Organization Osaka National Hospital, Japan, ２Department of Central Laboratory and Surgical Pathology, National Hospital Organization Osaka National Hospital, Japan and ３Department of Orthopaedic Surgery, National Hospital Organization Osaka National Hospital, Japan Received for publication June 30, 2014; Accepted for publication September 25, 2014 Correspondence to Dr. Yoshiko Okita, [email protected]
1297
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Discussion
Figure 1. Plain CT obtained at the time of admission. The plain CT scan revealed IVH primarily in the third ventricle without hydrocephalus.
the IVH. Neuroendoscopic surgery was subsequently performed. The initial examination showed slightly bloody cerebrospinal fluid (CSF). A hematoma was also observed in the third ventricle via the foramen of Monro, although it appeared to be difficult to remove owing to the presence of fibrous tissue. An endoscopic biopsy of the hematoma was therefore performed, and, after septostomy, an extraventricular drainage catheter was inserted into the left lateral ventricle. The patient’s headache, vomiting and somnolence diminished after the operation. T1-weighted magnetic resonance imaging (MRI) revealed a high-intensity mass, which corresponded to the hematoma seen on the CT scan (Fig. 2A), while T2-weighted MRI showed isointensity (Fig. 2B). The lesion measured 15 mm in diameter and was located in the third ventricle with an intact ventricular wall. Gadoliniumenhanced MRI disclosed a slightly enhanced mass, which was consequently found to be mostly occupied by the hematoma (Fig. 2C, D). A sagittal Fluid Attenuated Inversion Recovery (FLAIR) image revealed that the lesion was located close to the roof of the third ventricle (Fig. 2D). In addition, cerebral angiography was performed, and left vertebral angiography demonstrated a tumor stain at the choroid plexus in the third ventricle fed by the posterior medial choroidal artery; the tumor stain appeared throughout the arterial (Fig. 3A) and venous (Fig. 3B) phases. The pathological results showed that the lesion was metastatic FTC with positive immunostaining for thyroglobulin and thyroid transcription factor-1 (Fig. 4). A pathological examination revealed tumor tissue with a hematoma, suggesting intratumoral hemorrhage. CSF cytology specimens were negative for malignant cells. The patient was therefore diagnosed with CPM originating from FTC. After the diagnosis, he underwent stereotactic radiation therapy (SRT) at a dose of 33 Gy and was discharged two months later without neurological deficits. Eight months after CPM, he currently remains alive.
CPM lesions originating from thyroid carcinoma are extremely rare, with only five cases reported thus far (8-12). In this report, we described the sixth such case; all six cases are summarized in Table 1. Most of the patients exhibited solitary metastasis to the choroid plexus; three cases were of follicular carcinoma and the others were of papillary thyroid carcinoma. The histology was limited to well-differentiated and indolent thyroid carcinoma. In previous reports of CPM derived from thyroid carcinoma, the patients were diagnosed less than three years after the diagnosis of primary thyroid carcinoma. However, in the present case, the patient presented with CPM 13 years after receiving a primary diagnosis of thyroid cancer. Intratumoral hemorrhage occurred in two of the six cases, including the present case. Wasita et al. reported that, in their case, the tumor was initially not recognized because it was obscured by the focal IVH. One month later, following blood reabsorption, the tumor was identified and removed (11). In our case, CT revealed the IVH, and we planned endoscopic hematoma evacuation, which ultimately could not be performed. Consequently, based on the pathological findings of a biopsy of the hematoma, the IVH proved to be an intratumoral hemorrhage that had leaked into the CSF. The patient was ultimately diagnosed with CPM based on the findings of angiography, MRI, surgery and pathology, as well as the fact that, in cases in which the primary tumor spreads to the ventricles, the choroid plexus, the most highly vascular component of the ventricle system, is the favored site (13). To the best of our knowledge, only 36 cases of metastasis to the choroid plexus from extraneural primary sites have been reported in the literature overall (8-12, 14-37). Renal cell carcinoma (RCC) is the most frequent source of CPM (19-21, 24, 26-30, 33, 35), followed by thyroid carcinoma (8-12) (Table 2). On the other hand, metastases to the brain parenchyma are frequently reported to originate from lung, breast and colon cancers, whereas kidney and thyroid lesions account for only 5.3% and 0.9-1.5% of cases, respectively (Table 2) (1, 6, 12, 34). The higher incidence of CPM derived from RCC suggests that there may be a tropism of this tumor type for the ventricle, possibly due to the structural similarity of the kidney and choroid plexus, as both organs act as plasma-filtering systems (38). Similarly, the increased tendency of thyroid cancers to be a more common source of CPM than brain metastases suggests that thyroid cancer cells possess a biological affinity to the choroid plexus. RCCs are generally divided into rapidly or slowly progressive types based on their clinical manifestations. The rapid type is often fatal, causing death within a few years, while patients with the slow type may survive for more than 10 years. Interestingly, metastasis of RCC to the choroid plexus occurs most often in association with the slowly progressive type (11, 29). Similarly, indolent DTC may have a
1298
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Figure 2. MRI after the operation. (A) A T1-weighted image revealed a 15-mm high-intensity round mass in the third ventricle, which corresponded to the hematoma on the CT scan. (B) The lesion displayed isointensity on a T2-weighted image. (C) Gadolinium contrast enhancement showed the lesion to be only slightly enhanced, likely because it was mostly occupied by the hematoma. (D) A sagittal FLAIR image revealed that the lesion was located close to the roof of the third ventricle.
Figure 3. Angiography. Left vertebral angiograms disclosed a tumor at the choroid plexus in the third ventricle, fed by the posterior medial choroidal artery. The tumor stain (arrow) appeared from the arterial to the venous phase. Angiography also indicated the location of the choroid plexus (arrowheads) and foramen of Monro (thin arrow). (A) Arterial phase. (B) Venous phase.
higher tendency to metastasize to the choroid plexus during a slowly progressive disease course than in cases of lethal
anaplastic thyroid cancer. To date, the optimal management of CPM remains uncer-
1299
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Figure 4. Pathological findings. (A) Hematoxylin and Eosin staining demonstrated growth of the columnar epithelium consisting of large and small ductal structures of low nuclear grade in addition to a hemorrhagic area. (B, C) Immunohistochemical staining for thyroid transcription factor-1 (TTF1) (B) and thyroglobulin (C), immunohistochemical markers of thyroid tissue, confirmed the cell type to be follicular thyroid tumor cells. Table 1. Summary of Choroid Plexus Metastases from Thyroid Carcinoma. References
Age /Sex
#8
NA
Solitary or multiple NA
Histology papillary
Time from Hemorrhage diagnosis for primary lesion NA NA
Survival time after CPM NA
# 12
62/M
Solitary
follicular
-
first presented with CPM
# 11 #9
75/M 88/M
Multiple (2 sites) Solitary
papillary papillary
+ -
2 years 3 years
2 years 13 months
# 10
74/F
Solitary
follicular
-
first presented with CPM
alive for 14 months
follicular
+
13 years
still alive for 8 months
Present case 58/M Solitary NA: not applicable, CPM: choroid plexus metastasis
tain, as these lesions are extremely rare. A few previous reports have shown that surgical management of CPM originating from thyroid cancer results in better outcomes (11, 12). In addition, Siomin et al. reported that SRT presents a safe and viable primary treatment option for CPM of RCC, non-small cell lung cancer and esophageal cancer, with a survival time after SRT of 25.3±23.4 months (39). In the present case, the tumor was of small size and SRT was chosen as a less invasive treatment. Significantly, the patient
alive
continues to be alive more than eight months after SRT without complications or recurrence. Although there are very few data regarding the efficacy of SRT for CPM derived from thyroid cancer, we demonstrated that our patient received effective and safe treatment. In conclusion, we herein reported a case of solitary CPM originating from FTC associated with tumor hemorrhage 13 years after the primary diagnosis. While this is an extremely rare event, it appears that RCC and thyroid carcinoma (espe-
1300
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Table 2. Summary of 36 Cases of Matastasis to the Choroid Plexus. Primary lesion No. with CPM for CPM Kidney
14/36(38.9%)
Trigone 7
Metastatic site of choroid plexus Body 3rd vent 4th vent 5
Thyroid 6/36(16.7%) 2 1 Colon 4/36(11.1%) 1 Lung 3/36(8.3%) 2 Skin 3/36(8.3%) 2 Breast 2/36(5.6%) 2 Bladder 1/36(2.8%) 1 Esophagus 1/36(2.8%) Stomach 1/36(2.8%) 1 Lymphoma 1/36(2.8%) 1 Total 36 18 7 CPM: choroid plexus metastasis, NA: not applicable
1
Other
1
1 1
5.3 2 2 1
1
1
4
2
cially that of the differentiated and slowly progressive type) may have a higher tendency to metastasize to the choroid plexus owing to an as yet unknown molecular mechanism. In addition, IVH may be a marker of CPM in patients with DTC over the long clinical course of this disease. Therefore, it is important to biopsy hematomas in order to obtain a pathological diagnosis with careful consideration for the potential for CPM. The authors state that they have no Conflict of Interest (COI). Acknowledgement This work was supported by Grant-in-Aid for Scientific Research from the Ministry of Education, Science and Culture of Japan (No. 24791520 to Y.O.).
References 1. Sherman SI. Thyroid carcinoma. Lancet 361: 501-511, 2003. 2. Lang BH, Lo CY, Chan WF, Lam KY, Wan KY. Staging systems for papillary thyroid carcinoma: a review and comparison. Ann Surg 245: 366-378, 2007. 3. Lang BH, Lo CY, Chan WF, Lam KY, Wan KY. Prognostic factors in papillary and follicular thyroid carcinoma: their implications for cancer staging. Ann Surg Oncol 14: 730-738, 2007. 4. McConahey WM, Hay ID, Woolner LB, van Heerden JA, Taylor WF. Papillary thyroid cancer treated at the Mayo Clinic, 1946 through 1970: initial manifestations, pathologic findings, therapy, and outcome. Mayo Clin Proc 61: 978-996, 1986. 5. Biswal BM, Bal CS, Sandhu MS, Padhy AK, Rath GK. Management of intracranial metastases of differentiated carcinoma of thyroid. J Neurooncol 22: 77-81, 1994. 6. Chiu AC, Delpassand ES, Sherman SI. Prognosis and treatment of brain metastases in thyroid carcinoma. J Clin Endocrinol Metab 82: 3637-3642, 1997. 7. Hjiyiannakis P, Jefferies S, Harmer CL. Brain metastases in patients with differentiated thyroid carcinoma. Clin Oncol (R Coll Radiol) 8: 327-330, 1996. 8. Ferrer García JC, Merino Torres JF, Ponce Marco JL, Pinon Selles F. Unusual metastasis of differentiated thyroid carcinoma. An Med Interna 19: 579-582, 2002. 9. Heery CR, Engelhard HH, Slavin KV, Michals EA, Villano JL. Unusual CNS presentation of thyroid cancer. Clin Neurol Neurosurg 114: 1107-1109, 2012. 10. Kitagawa Y, Higuchi F, Abe Y, Matsuda H, Kim P, Ueki K. Metastasis to the choroid plexus from thyroid cancer: case report.
Frequency of all brain metastasis(%)
1.4 5.7 51.9 1.0 9.3 0.8 NA 4.8 0.7
References # 18-20, 23, 25-29, 32, 34 # 8-12 # 13, 24, 25, 27 # 21, 25, 36 # 14, 16, 22 # 15, 22 # 31 # 35 # 30 # 17
5
Neurol Med Chir (Tokyo) 53: 832-836, 2013. 11. Wasita B, Sakamoto M, Mizushima M, Kurosaki M, Watanabe T. Choroid plexus metastasis from papillary thyroid carcinoma presenting with intraventricular hemorrhage: case report. Neurosurgery 66: E1213-E1214, 2010. 12. Zhang YA, Kavar B, Drummond KJ. Thyroid carcinoma metastasis to the choroid plexus of the lateral ventricle. J Clin Neurosci 16: 118-121, 2009. 13. Sava I, Sava A, Sapte E, et al. Intraventricular metastatic clear cell renal carcinoma. Rom J Morphol Embryol 54: 447-450, 2013. 14. Al-Anazi A, Shannon P, Guha A. Solitary metastasis to the choroid plexus. Case illustration. J Neurosurg 92: 506, 2000. 15. Arbelaez A, Castillo M, Armao DM. Imaging features of intraventricular melanoma. AJNR Am J Neuroradiol 20: 691-693, 1999. 16. Della Puppa A, Dal Pos S, Zovato S, et al. Solitary intraventricular brain metastasis from a breast carcinoma. J Neurooncol 97: 123-126, 2010. 17. Escott EJ. A variety of appearances of malignant melanoma in the head: a review. Radiographics 21: 625-639, 2001. 18. Fukui K, Okamura K, Watanabe M, et al. Choroid plexus involvement in malignant lymphoma. Case report. Neurol Med Chir (Tokyo) 30: 869-873, 1990. 19. Hillard VH, Musunuru K, Hasan I, Zia S, Hirschfeld A. Longterm management of bilateral metastases of renal cell carcinoma to the choroid plexus. Acta Neurochir (Wien) 145: 793-797, 2003. 20. Iwatsuki K, Sato M, Taguchi J, et al. Choroid plexus metastasis of renal cell carcinoma causing intraventricular hemorrhage: a case report. No Shinkei Geka 27: 359-363, 1999 (in Japanese, Abstract in English). 21. Kadrian D, Tan L. Single choroid plexus metastasis 16 years after nephrectomy for renal cell carcinoma: case report and review of the literature. J Clin Neurosci 11: 88-91, 2004. 22. Kart BH, Reddy SC, Rao GR, Poveda H. Choroid plexus metastasis: CT appearance. J Comput Assist Tomogr 10: 537-540, 1986. 23. Kendall B, Reider-Grosswasser I, Valentine A. Diagnosis of masses presenting within the ventricles on computed tomography. Neuroradiology 25: 11-22, 1983. 24. Killebrew K, Krigman M, Mahaley MS Jr, Scatliff JH. Metastatic renal cell carcinoma mimicking a meningioma. Neurosurgery 13: 430-434, 1983. 25. Kitajima K, Morita M, Morikawa M, Sugimura K. Choroid plexus metastasis of colon cancer. Magn Reson Med Sci 2: 155-158, 2003. 26. Kohno M, Matsutani M, Sasaki T, Takakura K. Solitary metastasis to the choroid plexus of the lateral ventricle. Report of three cases and a review of the literature. J Neurooncol 27: 47-52, 1996. 27. Lauretti L, Fernandez E, Pallini R, et al. Long survival in an untreated solitary choroid plexus metastasis from renal cell carci-
1301
Intern Med 54: 1297-1302, 2015
28.
29.
30.
31.
32.
33.
DOI: 10.2169/internalmedicine.54.3560
noma: case report and review of the literature. J Neurooncol 71: 157-160, 2005. Leach JC, Garrott H, King JA, Kaye AH. Solitary metastasis to the choroid plexus of the third ventricle mimicking a colloid cyst: a report of two cases. J Clin Neurosci 11: 521-523, 2004. Matsumura H, Yoshimine T, Yamamoto S, et al. Single solitary metastasis of the slowly progressive type of renal cell carcinoma to the choroid plexus--case report. Neurol Med Chir (Tokyo) 37: 916-919, 1997. Mizuno M, Asakura K, Nakajima S, et al. Renal cell carcinoma metastasizing to choroid plexus of lateral ventricle; a case report. No Shinkei Geka 20: 469-474, 1992 (in Japanese, Abstract in English). Nakabayashi H, Murata K, Sakaguchi M, Nakajima K, Katsuyama J. Choroid plexus metastasis from gastric cancer--case report. Neurol Med Chir (Tokyo) 34: 183-186, 1994. Qasho R, Tommaso V, Rocchi G, Simi U, Delfini R. Choroid plexus metastasis from carcinoma of the bladder: case report and review of the literature. J Neurooncol 45: 237-240, 1999. Raila FA, Bottoms WT Jr, Fratkin JD. Solitary choroid plexus metastasis from a renal cell carcinoma. South Med J 91: 1159-1162,
1998. 34. Schouten LJ, Rutten J, Huveneers HA, Twijnstra A. Incidence of brain metastases in a cohort of patients with carcinoma of the breast, colon, kidney, and lung and melanoma. Cancer 94: 26982705, 2002. 35. Spetzger U, Mull M, Sure U, Gilsbach J. Subarachnoid and intraventricular hemorrhage caused by hypernephroma metastasis, accompanied by innocent bilateral posterior communicating artery aneurysms. Surg Neurol 44: 275-278, 1995. 36. Sung WS, Dubey A, Erasmus A, Hunn A. Solitary choroid plexus metastasis from carcinoma of the oesophagus. J Clin Neurosci 15: 594-597, 2008. 37. Tanimoto M, Tatsumi S, Tominaga S, et al. Choroid plexus metastasis of lung carcinoma--case report. Neurol Med Chir (Tokyo) 31: 152-155, 1991. 38. Vecil GG, Lang FF. Surgical resection of metastatic intraventricular tumors. Neurosurg Clin N Am 14: 593-606, 2003. 39. Siomin V, Lin JL, Marko NF, et al. Stereotactic radiosurgical treatment of brain metastases to the choroid plexus. Int J Radiat Oncol Biol Phys 80: 1134-1142, 2011.
Ⓒ 2015 The Japanese Society of Internal Medicine http://www.naika.or.jp/imonline/index.html
1302
CASE REPORT
□
Choroid Plexus Metastasis of Follicular Thyroid Carcinoma Diagnosed due to Intraventricular Hemorrhage Toru Umehara 1, Yoshiko Okita 1, Masahiro Nonaka 1, Kosuke Mori 1, Yonehiro Kanemura 1, Yoshinori Kodama 2, Masayuki Mano 2, Ikuo Kudawara 3 and Shin Nakajima 1
Abstract Choroid plexus metastasis (CPM) is extremely rare and originates most frequently from renal cell carcinoma (RCC). We herein report the case of a 58-year-old man who developed a solitary CPM lesion derived from follicular thyroid carcinoma in addition to intraventricular hemorrhage. Computed tomography revealed acute hydrocephalus as a result of the hemorrhage, and we planned endoscopic hematoma evacuation. Since it was too difficult to reach the hematoma, we considered the possibility of a neoplasm and performed a biopsy of the lesion, the results of which led to an accurate diagnosis of CPM in this case. We also review previous reports of CPM originating from thyroid carcinoma compared with RCC. Key words: intraventricular hemorrhage, choroid plexus metastasis, follicular thyroid carcinoma (Intern Med 54: 1297-1302, 2015) (DOI: 10.2169/internalmedicine.54.3560)
we describe the sixth case of FTC metastasizing to the choroid plexus in a patient presenting with intraventricular hemorrhage (IVH) 13 years after total thyroidectomy.
Introduction The incidence of thyroid carcinoma is fairly low, accounting for only approximately 1% of new cases of malignant disease worldwide. The majority (94%) of such cases involve differentiated thyroid carcinoma (DTC), either papillary thyroid carcinoma or follicular thyroid carcinoma (FTC) (1), which is generally associated with an indolent disease course. Indeed, the prognosis of DTC remains excellent, with the 10-year cancer-specific survival rate reaching 90% (2, 3). Local invasion is the usual mode of spread, followed by dissemination via the lymphatic or hematogenous route. The lungs and bone are the most common sites of distant metastasis. Conversely, brain metastasis of DTC is very rare, occurring in approximately 0.9% of the patient population, and is a marker of a poor prognosis (4). The disease-specific mortality rate of DTC metastasis is 67%, with a median product-limit survival of 12.4 months (4-7). In addition, among types of brain metastasis cases, choroid plexus metastasis (CPM) is extremely rare, with only five cases reported in the literature to date (8-12). In this report,
Case Report We herein report the case of a 58-year-old man who presented with severe headache, vomiting and somnolence. He had undergone total thyroidectomy for FTC 13 years prior to presentation and had received radiation therapy for multiple bone metastases in the right clavicle, right fifth rib and lumbar vertebrae (L2-3). A clinical examination performed on admission revealed a Glasgow coma scale score of 14 points (E4, V4, M6), with no focal neurological deficits. A computed tomography (CT) scan of the head showed IVH in the third ventricle without hydrocephalus (Fig. 1), whereas subsequent three-dimensional CT angiography (CTA) revealed no evidence of intracranial vascular abnormalities, initially suspected to be the source of bleeding. Half a day after presentation, the patient exhibited slightly disturbed consciousness resulting from hydrocephalus. Accordingly, we planned endoscopic hematoma evacuation for
１
Department of Neurosurgery, National Hospital Organization Osaka National Hospital, Japan, ２Department of Central Laboratory and Surgical Pathology, National Hospital Organization Osaka National Hospital, Japan and ３Department of Orthopaedic Surgery, National Hospital Organization Osaka National Hospital, Japan Received for publication June 30, 2014; Accepted for publication September 25, 2014 Correspondence to Dr. Yoshiko Okita, [email protected]
1297
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Discussion
Figure 1. Plain CT obtained at the time of admission. The plain CT scan revealed IVH primarily in the third ventricle without hydrocephalus.
the IVH. Neuroendoscopic surgery was subsequently performed. The initial examination showed slightly bloody cerebrospinal fluid (CSF). A hematoma was also observed in the third ventricle via the foramen of Monro, although it appeared to be difficult to remove owing to the presence of fibrous tissue. An endoscopic biopsy of the hematoma was therefore performed, and, after septostomy, an extraventricular drainage catheter was inserted into the left lateral ventricle. The patient’s headache, vomiting and somnolence diminished after the operation. T1-weighted magnetic resonance imaging (MRI) revealed a high-intensity mass, which corresponded to the hematoma seen on the CT scan (Fig. 2A), while T2-weighted MRI showed isointensity (Fig. 2B). The lesion measured 15 mm in diameter and was located in the third ventricle with an intact ventricular wall. Gadoliniumenhanced MRI disclosed a slightly enhanced mass, which was consequently found to be mostly occupied by the hematoma (Fig. 2C, D). A sagittal Fluid Attenuated Inversion Recovery (FLAIR) image revealed that the lesion was located close to the roof of the third ventricle (Fig. 2D). In addition, cerebral angiography was performed, and left vertebral angiography demonstrated a tumor stain at the choroid plexus in the third ventricle fed by the posterior medial choroidal artery; the tumor stain appeared throughout the arterial (Fig. 3A) and venous (Fig. 3B) phases. The pathological results showed that the lesion was metastatic FTC with positive immunostaining for thyroglobulin and thyroid transcription factor-1 (Fig. 4). A pathological examination revealed tumor tissue with a hematoma, suggesting intratumoral hemorrhage. CSF cytology specimens were negative for malignant cells. The patient was therefore diagnosed with CPM originating from FTC. After the diagnosis, he underwent stereotactic radiation therapy (SRT) at a dose of 33 Gy and was discharged two months later without neurological deficits. Eight months after CPM, he currently remains alive.
CPM lesions originating from thyroid carcinoma are extremely rare, with only five cases reported thus far (8-12). In this report, we described the sixth such case; all six cases are summarized in Table 1. Most of the patients exhibited solitary metastasis to the choroid plexus; three cases were of follicular carcinoma and the others were of papillary thyroid carcinoma. The histology was limited to well-differentiated and indolent thyroid carcinoma. In previous reports of CPM derived from thyroid carcinoma, the patients were diagnosed less than three years after the diagnosis of primary thyroid carcinoma. However, in the present case, the patient presented with CPM 13 years after receiving a primary diagnosis of thyroid cancer. Intratumoral hemorrhage occurred in two of the six cases, including the present case. Wasita et al. reported that, in their case, the tumor was initially not recognized because it was obscured by the focal IVH. One month later, following blood reabsorption, the tumor was identified and removed (11). In our case, CT revealed the IVH, and we planned endoscopic hematoma evacuation, which ultimately could not be performed. Consequently, based on the pathological findings of a biopsy of the hematoma, the IVH proved to be an intratumoral hemorrhage that had leaked into the CSF. The patient was ultimately diagnosed with CPM based on the findings of angiography, MRI, surgery and pathology, as well as the fact that, in cases in which the primary tumor spreads to the ventricles, the choroid plexus, the most highly vascular component of the ventricle system, is the favored site (13). To the best of our knowledge, only 36 cases of metastasis to the choroid plexus from extraneural primary sites have been reported in the literature overall (8-12, 14-37). Renal cell carcinoma (RCC) is the most frequent source of CPM (19-21, 24, 26-30, 33, 35), followed by thyroid carcinoma (8-12) (Table 2). On the other hand, metastases to the brain parenchyma are frequently reported to originate from lung, breast and colon cancers, whereas kidney and thyroid lesions account for only 5.3% and 0.9-1.5% of cases, respectively (Table 2) (1, 6, 12, 34). The higher incidence of CPM derived from RCC suggests that there may be a tropism of this tumor type for the ventricle, possibly due to the structural similarity of the kidney and choroid plexus, as both organs act as plasma-filtering systems (38). Similarly, the increased tendency of thyroid cancers to be a more common source of CPM than brain metastases suggests that thyroid cancer cells possess a biological affinity to the choroid plexus. RCCs are generally divided into rapidly or slowly progressive types based on their clinical manifestations. The rapid type is often fatal, causing death within a few years, while patients with the slow type may survive for more than 10 years. Interestingly, metastasis of RCC to the choroid plexus occurs most often in association with the slowly progressive type (11, 29). Similarly, indolent DTC may have a
1298
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Figure 2. MRI after the operation. (A) A T1-weighted image revealed a 15-mm high-intensity round mass in the third ventricle, which corresponded to the hematoma on the CT scan. (B) The lesion displayed isointensity on a T2-weighted image. (C) Gadolinium contrast enhancement showed the lesion to be only slightly enhanced, likely because it was mostly occupied by the hematoma. (D) A sagittal FLAIR image revealed that the lesion was located close to the roof of the third ventricle.
Figure 3. Angiography. Left vertebral angiograms disclosed a tumor at the choroid plexus in the third ventricle, fed by the posterior medial choroidal artery. The tumor stain (arrow) appeared from the arterial to the venous phase. Angiography also indicated the location of the choroid plexus (arrowheads) and foramen of Monro (thin arrow). (A) Arterial phase. (B) Venous phase.
higher tendency to metastasize to the choroid plexus during a slowly progressive disease course than in cases of lethal
anaplastic thyroid cancer. To date, the optimal management of CPM remains uncer-
1299
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Figure 4. Pathological findings. (A) Hematoxylin and Eosin staining demonstrated growth of the columnar epithelium consisting of large and small ductal structures of low nuclear grade in addition to a hemorrhagic area. (B, C) Immunohistochemical staining for thyroid transcription factor-1 (TTF1) (B) and thyroglobulin (C), immunohistochemical markers of thyroid tissue, confirmed the cell type to be follicular thyroid tumor cells. Table 1. Summary of Choroid Plexus Metastases from Thyroid Carcinoma. References
Age /Sex
#8
NA
Solitary or multiple NA
Histology papillary
Time from Hemorrhage diagnosis for primary lesion NA NA
Survival time after CPM NA
# 12
62/M
Solitary
follicular
-
first presented with CPM
# 11 #9
75/M 88/M
Multiple (2 sites) Solitary
papillary papillary
+ -
2 years 3 years
2 years 13 months
# 10
74/F
Solitary
follicular
-
first presented with CPM
alive for 14 months
follicular
+
13 years
still alive for 8 months
Present case 58/M Solitary NA: not applicable, CPM: choroid plexus metastasis
tain, as these lesions are extremely rare. A few previous reports have shown that surgical management of CPM originating from thyroid cancer results in better outcomes (11, 12). In addition, Siomin et al. reported that SRT presents a safe and viable primary treatment option for CPM of RCC, non-small cell lung cancer and esophageal cancer, with a survival time after SRT of 25.3±23.4 months (39). In the present case, the tumor was of small size and SRT was chosen as a less invasive treatment. Significantly, the patient
alive
continues to be alive more than eight months after SRT without complications or recurrence. Although there are very few data regarding the efficacy of SRT for CPM derived from thyroid cancer, we demonstrated that our patient received effective and safe treatment. In conclusion, we herein reported a case of solitary CPM originating from FTC associated with tumor hemorrhage 13 years after the primary diagnosis. While this is an extremely rare event, it appears that RCC and thyroid carcinoma (espe-
1300
Intern Med 54: 1297-1302, 2015
DOI: 10.2169/internalmedicine.54.3560
Table 2. Summary of 36 Cases of Matastasis to the Choroid Plexus. Primary lesion No. with CPM for CPM Kidney
14/36(38.9%)
Trigone 7
Metastatic site of choroid plexus Body 3rd vent 4th vent 5
Thyroid 6/36(16.7%) 2 1 Colon 4/36(11.1%) 1 Lung 3/36(8.3%) 2 Skin 3/36(8.3%) 2 Breast 2/36(5.6%) 2 Bladder 1/36(2.8%) 1 Esophagus 1/36(2.8%) Stomach 1/36(2.8%) 1 Lymphoma 1/36(2.8%) 1 Total 36 18 7 CPM: choroid plexus metastasis, NA: not applicable
1
Other
1
1 1
5.3 2 2 1
1
1
4
2
cially that of the differentiated and slowly progressive type) may have a higher tendency to metastasize to the choroid plexus owing to an as yet unknown molecular mechanism. In addition, IVH may be a marker of CPM in patients with DTC over the long clinical course of this disease. Therefore, it is important to biopsy hematomas in order to obtain a pathological diagnosis with careful consideration for the potential for CPM. The authors state that they have no Conflict of Interest (COI). Acknowledgement This work was supported by Grant-in-Aid for Scientific Research from the Ministry of Education, Science and Culture of Japan (No. 24791520 to Y.O.).
References 1. Sherman SI. Thyroid carcinoma. Lancet 361: 501-511, 2003. 2. Lang BH, Lo CY, Chan WF, Lam KY, Wan KY. Staging systems for papillary thyroid carcinoma: a review and comparison. Ann Surg 245: 366-378, 2007. 3. Lang BH, Lo CY, Chan WF, Lam KY, Wan KY. Prognostic factors in papillary and follicular thyroid carcinoma: their implications for cancer staging. Ann Surg Oncol 14: 730-738, 2007. 4. McConahey WM, Hay ID, Woolner LB, van Heerden JA, Taylor WF. Papillary thyroid cancer treated at the Mayo Clinic, 1946 through 1970: initial manifestations, pathologic findings, therapy, and outcome. Mayo Clin Proc 61: 978-996, 1986. 5. Biswal BM, Bal CS, Sandhu MS, Padhy AK, Rath GK. Management of intracranial metastases of differentiated carcinoma of thyroid. J Neurooncol 22: 77-81, 1994. 6. Chiu AC, Delpassand ES, Sherman SI. Prognosis and treatment of brain metastases in thyroid carcinoma. J Clin Endocrinol Metab 82: 3637-3642, 1997. 7. Hjiyiannakis P, Jefferies S, Harmer CL. Brain metastases in patients with differentiated thyroid carcinoma. Clin Oncol (R Coll Radiol) 8: 327-330, 1996. 8. Ferrer García JC, Merino Torres JF, Ponce Marco JL, Pinon Selles F. Unusual metastasis of differentiated thyroid carcinoma. An Med Interna 19: 579-582, 2002. 9. Heery CR, Engelhard HH, Slavin KV, Michals EA, Villano JL. Unusual CNS presentation of thyroid cancer. Clin Neurol Neurosurg 114: 1107-1109, 2012. 10. Kitagawa Y, Higuchi F, Abe Y, Matsuda H, Kim P, Ueki K. Metastasis to the choroid plexus from thyroid cancer: case report.
Frequency of all brain metastasis(%)
1.4 5.7 51.9 1.0 9.3 0.8 NA 4.8 0.7
References # 18-20, 23, 25-29, 32, 34 # 8-12 # 13, 24, 25, 27 # 21, 25, 36 # 14, 16, 22 # 15, 22 # 31 # 35 # 30 # 17
5
Neurol Med Chir (Tokyo) 53: 832-836, 2013. 11. Wasita B, Sakamoto M, Mizushima M, Kurosaki M, Watanabe T. Choroid plexus metastasis from papillary thyroid carcinoma presenting with intraventricular hemorrhage: case report. Neurosurgery 66: E1213-E1214, 2010. 12. Zhang YA, Kavar B, Drummond KJ. Thyroid carcinoma metastasis to the choroid plexus of the lateral ventricle. J Clin Neurosci 16: 118-121, 2009. 13. Sava I, Sava A, Sapte E, et al. Intraventricular metastatic clear cell renal carcinoma. Rom J Morphol Embryol 54: 447-450, 2013. 14. Al-Anazi A, Shannon P, Guha A. Solitary metastasis to the choroid plexus. Case illustration. J Neurosurg 92: 506, 2000. 15. Arbelaez A, Castillo M, Armao DM. Imaging features of intraventricular melanoma. AJNR Am J Neuroradiol 20: 691-693, 1999. 16. Della Puppa A, Dal Pos S, Zovato S, et al. Solitary intraventricular brain metastasis from a breast carcinoma. J Neurooncol 97: 123-126, 2010. 17. Escott EJ. A variety of appearances of malignant melanoma in the head: a review. Radiographics 21: 625-639, 2001. 18. Fukui K, Okamura K, Watanabe M, et al. Choroid plexus involvement in malignant lymphoma. Case report. Neurol Med Chir (Tokyo) 30: 869-873, 1990. 19. Hillard VH, Musunuru K, Hasan I, Zia S, Hirschfeld A. Longterm management of bilateral metastases of renal cell carcinoma to the choroid plexus. Acta Neurochir (Wien) 145: 793-797, 2003. 20. Iwatsuki K, Sato M, Taguchi J, et al. Choroid plexus metastasis of renal cell carcinoma causing intraventricular hemorrhage: a case report. No Shinkei Geka 27: 359-363, 1999 (in Japanese, Abstract in English). 21. Kadrian D, Tan L. Single choroid plexus metastasis 16 years after nephrectomy for renal cell carcinoma: case report and review of the literature. J Clin Neurosci 11: 88-91, 2004. 22. Kart BH, Reddy SC, Rao GR, Poveda H. Choroid plexus metastasis: CT appearance. J Comput Assist Tomogr 10: 537-540, 1986. 23. Kendall B, Reider-Grosswasser I, Valentine A. Diagnosis of masses presenting within the ventricles on computed tomography. Neuroradiology 25: 11-22, 1983. 24. Killebrew K, Krigman M, Mahaley MS Jr, Scatliff JH. Metastatic renal cell carcinoma mimicking a meningioma. Neurosurgery 13: 430-434, 1983. 25. Kitajima K, Morita M, Morikawa M, Sugimura K. Choroid plexus metastasis of colon cancer. Magn Reson Med Sci 2: 155-158, 2003. 26. Kohno M, Matsutani M, Sasaki T, Takakura K. Solitary metastasis to the choroid plexus of the lateral ventricle. Report of three cases and a review of the literature. J Neurooncol 27: 47-52, 1996. 27. Lauretti L, Fernandez E, Pallini R, et al. Long survival in an untreated solitary choroid plexus metastasis from renal cell carci-
1301
Intern Med 54: 1297-1302, 2015
28.
29.
30.
31.
32.
33.
DOI: 10.2169/internalmedicine.54.3560
noma: case report and review of the literature. J Neurooncol 71: 157-160, 2005. Leach JC, Garrott H, King JA, Kaye AH. Solitary metastasis to the choroid plexus of the third ventricle mimicking a colloid cyst: a report of two cases. J Clin Neurosci 11: 521-523, 2004. Matsumura H, Yoshimine T, Yamamoto S, et al. Single solitary metastasis of the slowly progressive type of renal cell carcinoma to the choroid plexus--case report. Neurol Med Chir (Tokyo) 37: 916-919, 1997. Mizuno M, Asakura K, Nakajima S, et al. Renal cell carcinoma metastasizing to choroid plexus of lateral ventricle; a case report. No Shinkei Geka 20: 469-474, 1992 (in Japanese, Abstract in English). Nakabayashi H, Murata K, Sakaguchi M, Nakajima K, Katsuyama J. Choroid plexus metastasis from gastric cancer--case report. Neurol Med Chir (Tokyo) 34: 183-186, 1994. Qasho R, Tommaso V, Rocchi G, Simi U, Delfini R. Choroid plexus metastasis from carcinoma of the bladder: case report and review of the literature. J Neurooncol 45: 237-240, 1999. Raila FA, Bottoms WT Jr, Fratkin JD. Solitary choroid plexus metastasis from a renal cell carcinoma. South Med J 91: 1159-1162,
1998. 34. Schouten LJ, Rutten J, Huveneers HA, Twijnstra A. Incidence of brain metastases in a cohort of patients with carcinoma of the breast, colon, kidney, and lung and melanoma. Cancer 94: 26982705, 2002. 35. Spetzger U, Mull M, Sure U, Gilsbach J. Subarachnoid and intraventricular hemorrhage caused by hypernephroma metastasis, accompanied by innocent bilateral posterior communicating artery aneurysms. Surg Neurol 44: 275-278, 1995. 36. Sung WS, Dubey A, Erasmus A, Hunn A. Solitary choroid plexus metastasis from carcinoma of the oesophagus. J Clin Neurosci 15: 594-597, 2008. 37. Tanimoto M, Tatsumi S, Tominaga S, et al. Choroid plexus metastasis of lung carcinoma--case report. Neurol Med Chir (Tokyo) 31: 152-155, 1991. 38. Vecil GG, Lang FF. Surgical resection of metastatic intraventricular tumors. Neurosurg Clin N Am 14: 593-606, 2003. 39. Siomin V, Lin JL, Marko NF, et al. Stereotactic radiosurgical treatment of brain metastases to the choroid plexus. Int J Radiat Oncol Biol Phys 80: 1134-1142, 2011.
Ⓒ 2015 The Japanese Society of Internal Medicine http://www.naika.or.jp/imonline/index.html
1302
