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A case of adenocarcinoma metastasis in meningioma mimicking cystic mass

�nraduction:

Metastasis of an extra-cranial tumor in an intra-cranial tumor is very rare [1]. Harboring intra-cranial tumor is frequently a meningioma while the most common metastasizing tumor is breast or lung cancer [2,3]. It is difficult to differentiate mass lesions accompanying the cyst from intra-axial primary glial neoplasms [4,5]. We presented an adenocarcinoma metastasis in meningioma, which mimicked a cystic mass.

Key Words: Metastasis, meningioma, Adenocarcinoma

Case:
A 61-year old male patient presented to the Emergency Service with loss of consciousness. He had no known systemic disease and history of malignancy. Neurological examination revealed grade 4/5 right hemiparesis. Cranial MR depicted a lesion with cyctic and solid components in the left frontal lobe (Fig_1a, b). The mass with extra-axial solid and intra-axial cystic component that invaded the dura was excised totally with left fronto-temporal craniotomy. Histopathological examination�showed the presence of metastatic adenocarcinoma within the fibrous type meningioma (figure 2).���Immunohistochemically, adenocarcinoma cells were positive for��pancytokeratin, cytokeratin 7, �TTF1 and�EMA,� and�� negative for cytokeratin 20, thyroglobulin, PSA, CD10 and CD30.� Histochemically adenocarcinoma cells were also positive for periodic acid-Schiff and alcian blue focally. Meningioma cells were positive for vimentin and EMA but negative for�pancytokeratin, cytokeratin 7 and�TTF1.� These findings were consistent with "metastatic adenocarcinoma and meningioma".� Further work-up for examination of lungs advised for confirmation. Thoracic CT did not show active infectious or neoplastic infiltration within the pulmonary parenchyma. PET depicted increased 18 FDG uptake in the inferior lobe of the left lung and left hilar lymph nodes. 

Discussion:
       The most common intratumoral metastasis in brain is in meningiomas [1,2]. Metastasis in meningioma, which is a primary tumor, is usually from breast (50%) or lung cancer (30%) though genitourinary tumors, lymphoma, renal cell cancer, prostatic cancer, gallbladder, melanoma, parotid tumor, multiple myeloma have also been reported to metastasize [1,6]. The present case involved adenocarcicoma metastasis in fibrous type meningioma in the frontal region though metastasis in all types of histological types and in all anatomical localizations have been reported [6,7]. Primary predisposing factors include loss of tumor suppressor gene, hormonal influences, especially in breast cancer metastasis, high vascularity of the meningioma, and lesser immune surveillance of the tumor tissue than normal tissue [3]. High collagen and lipid content of the meningioma makes it an ideal environment for the tumor cells to adhere [11]. Moreover, increased E-Catedrin, a surface adhesion molecule in meningiomas may be a factor in the metastasis of tumor cells in here [8]. 
    Preoperative diagnosis of metastasis in a meningioma is very difficult [1]. Even though there was a debate with regard to the preoperative radiologic diagnosis of our patient with adenocarcinoma metastasis in meningioma particularly since it was accompanied by a cyst and there was no history of malignancy, histopathology was definitive. When suspected MR spectroscopy, can be utilized for differential diagnosis [6].
   Cystic meningioma refers to meningiomas with intratumoral or peritumoral cyst [4]. Large cystic meningiomas that present with atypical features, as was the case in our patient, are more common male and pediatric patients [10]. This can lead to misdiagnosis such as cystic or necrotic glioma. Numerous mechanisms have been proposed to explain cyst formation in cystic meningioma, which include cystic degeneration of the tumor, secretion of fluid by the tumor cells, diapedesis of cerebrospinal fluid (CSF) into the scar tissue and sequestration of CSF by neighboring tumor [4,10]. We believe that the fact that the metastasizing tumor was adenocarcinoma might have contributed to the development of a cyst in a meningioma. Nauta and coworkers classified cystic meningiomas into 4 groups; centrally-located intratumoral cyst, peripherally-located intratumoral cyst, peritumoral cyst within the neighboring parenchyma and peritumoral cyst between the tumor and neighboring parenchyma, the type in the present case [11]. Metastasis in meningioma should be included in differential diagnosis if there is sudden deterioration in the health of patients known to have a meningioma or if there is rapid increase in the size of the tumor in patients with a history of cancer [12]. Metastasis in meningioma should be suspected in the presence of an atypical radiological appearance or an extra-axial tumor with a large cyst. In the presence of meningioma with metastasis while there is no history of a primary tumor, especially lung or breast cancer should be kept in mind in males and females, respectively, and systemic cancer screening should be carried out. 

CONCLUSION
Metastasis in meningioma should be considered in differential diagnosis when intra-axial masses accompanied by a cyst cannot be differentiated from extra-axial masses accompanied by a cyst. 

























REFERENCES:

1. Lanotte M, Benech F, Panciani PP, Cassoni P, Ducati A (2009) Systemic cancer metastasis in a meningioma: report of two cases and review of the literature.Clin Neurol Neurosurg 111(1):87-93.

2. Schmitt HP (1984) Metastases of malignant neoplasms to intracranial tumours: the �tumour-in-a-tumour� phenomenon. Virchows Arch 405:155�160

3.  Lee A, Wallace C, Rewcastle B, Sutherland G.Metastases to meningioma (1998) AJNR Am J Neuroradiol 19(6):1120-2.

4. Buetow MP, Buetow PC, Smirniotopoulos JG (1991) Typical, atypical, and misleading features in meningioma. Radiographics 11(6):1087�1106.

5. Wolintz AH, Mastri A (1970) Metastasis of carcinoma of lung to sphenoid ridge of meningioma. NY State J Med 70:2592-2598.

6. Widdel L, Kleinschmidt-Demasters BK, Kindt G (2010) Tumor-to-Tumor Metastasis from Hematopoietic Neoplasms to Meningiomas: Report of Two Patients with Significant Cerebral Edema. World Neurosurg 74(1):165-71. 

7. Aghi M, Kiehl TR, Brisman JL (2005) Breast adenocarcinoma metastatic to epidural cervical spine meningioma: case report and review of the literature. J Neurooncol 75:149-155. 

8. Figarella-Branger D, Pellisier JF, Bouillot P, Graziani N, Gambarelli D,  et al. (1994) Expression of neural cell-adhesion molecule isoforms and epithelial cadherin adhesion molecules in 47 human meningiomas: correlation with clinical and morphological data. Mod Pathol 7:752-761.


9. Shiino A, Nakasu S, Matsuda M, Handa J, Morikawa S, et al. (1999)Noninvasive evaluation of the malignant potential of intracranial meningiomas performed using proton magnetic resonance spectroscopy. J Neurosurg 91: 928�34.

10. Jung TY, Jung S, Shin SR, Moon KS, Kim IY, et al. (2005) Clinical and histopathological analysis of cystic meningiomas. J Clin Neurosci. 12(6):651-5.

11. Nauta HJ, Tucker WS, Horsey WJ, Gonsalves C. (1979) Xanthochromic cysts associated with meningioma. J Neurol Neurosurg Psychiatry 42:529�35.

12. Kim KH, Hong EK, Lee SH, Yoo H. (2013) Non small cell carcinoma metastasis to meningioma. J Korean Neurosurg Soc. 53(1):43-5.





Fig_1a: Axial T1; Mass and peritumoral cyst, which displaces the midline structures 1b: Axial contrast-enhanced T1-weighted image depicts marked enhancement of the solid component and contrast-enhanced septation around the cystic component. 




Fig_2: Histopathologic�examination revealed fibrous meningioma and�metastatic adenocarcinoma�in the same area. A psammoma body was�also seen in�the meningioma.



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