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Surprise within a meningioma: case report of signet ring cell carcinoma metastasis in a meningioma We present a 66-year-old lady who complained of left-sided headaches in the 3 weeks prior to presentation. Magnetic resonance imaging (MRI) scan showed features consistent with a left sphenoid wing meningioma (Fig. 1). She underwent a left pterional craniotomy. A hyperostotic, haemorrhagic sphenoid ridge was encountered along with multiple small dural feeding vessels. The tumour was soft, friable and haemorrhagic. Simpson II resection was achieved. Post-operative neurological recovery was uneventful. Histopathology showed uniform meningothelial cells with no atypical features, corresponding with WHO Grade I. There were also aggregates of discohesive signet ring cells seen percolating through patchy areas of the tumour. These cells showed strong immunoreactivity for CK20 and variable positivity for CK7. CEA and BerEP4 highlighted signet ring cells (Fig. 2). They were negative for ER, TTF-1, CD15 and S100. The findings favoured a metastasis from the upper gastrointestinal or genitourinary tracts, with the

diagnosis of exclusion being a metaplastic meningioma with mucinous differentiation. A search was commenced to locate the primary site. Gastroscopy and biopsy confirmed signet ring cell carcinoma at the gastrooesophageal junction (Fig. 3). Positron emission tomography scan showed no other metastases. Retrospectively, the patient had complained of dysphagia and weight loss in the preceding 2 months. Radiotherapy was commenced, but this lady’s condition deteriorated rapidly over the subsequent 4 months. The last scan prior to her death showed no recurrence or further metastasis in her brain. Meningiomas, while mostly benign in nature, can harbour malignant metastases that originate from another primary tumour type.1 Intrameningioma metastatic tumours have origins from various parts of the body, but the most common donors are breast and lung tumours.1–3 Other rare cases of metastasis from primary tumours have also been reported, including prostate adenocarcinoma, renal

Fig. 1. Left sphenoid wing meningioma. Slightly hyperintense on axial T2/FLAIR sequence magnetic resonance imaging (left) with signal abnormality extending posteriorly to the peritrigonal white matter. It enhances avidly post contrast (right) with an enhancing dural tail.

Fig. 2. Histopathology of intrameningioma signet ring cells metastasis. Aggregates of signet ring cells percolating through the meningioma seen on right upper corner of this hematoxylin and eosin stained section (original magnification 20×) (left). Immunohistochemical stain for carcinoembryonic antigen highlighting the signet ring cells (original magnification 20×) (right).

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Fig. 3. Gastroscopy of lower third of oesophagus. Non-obstructing, circumferential mass measuring 5 cm in length.

carcinoma, haematopoietic cancers, melanoma and genitourinary tumours.2 We present a case of an intrameningioma metastatic tumour that had its origin in the gastrointestinal system as evidenced by the presence of signet ring cells. The only other case report that is similar to this report is that by Honma et al. in 1989 who observed the presence of gastric carcinoma metastasis within a meningioma on autopsy of an elderly Japanese lady.3 Meningiomas are the third most frequent recipient tumour type2 and the most common intracranial neoplasm to harbour metastases.1,4 Among the hypotheses for this observation is the rich vascularity of meningiomas that create an easy route for haematogenous spread of malignant tumours.1,2,5 Other proposals include the noncompetitive environment within a meningioma accorded to metastasis due to its slow metabolic rate, relatively unrestricted tumour proliferation from the lack of immune support within meningiomas and the high lipid and collagen content within meningiomas that provide a source of nutrients for the metastatic tumour.1,2 It is also worth noting that all histologic types of meningiomas from all anatomic locations are equally likely to host metastatic tumours.2 There are key points that can be learned from this case. Firstly, a high index of suspicion is essential to remind the clinician of the possibility of a metastatic tumour hosted within highly vascularized tumours such as meningiomas, especially if the patient also presents with other non-specific and/or non-neurological symptoms. Conventional computed tomography and MRI has its limitations and cannot reliably determine the presence of metastasis within a meningioma.1,5 Hence, it also highlights the significant importance of histopathology and the role played by the pathologist not only in classifying the grade of meningioma but more crucially in determining the presence of a metastatic tumour type within it. The finding of an intrameningioma metastasis should place an obligation on the treating team for a thorough systemic investigation in search of the other primary tumour and any of its metastases. Coexistence of two separate benign components has been reported in meningiomas,6 but one has to rule out all other options before

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diagnosing this rare occurrence or metaplastic meningioma. Although the initial neurological complaints may be largely resolved through surgical resection of the meningioma, a proven intrameningioma metastatic tumour changes the outlook of a curable tumour into one with a poor overall prognosis. With this in mind, a realistic management plan has to be formulated to address the patient’s expectations. Various authors have supported the use of relatively new cuttingedge neuroimaging methods such as perfusion MRI and proton magnetic resonance spectroscopy to help identify the presence of metastasis within a meningioma.1,5,7 However, the indications for use of these new technologies are still debatable and do not replace histopathology as the only reliable method for diagnosing this unique occurrence.

References 1. Moody P, Murtagh K, Piduru S, Brem S, Murtagh R, Rojiani AM. Tumorto-tumor metastasis: pathology and neuroimaging considerations. Int. J. Clin. Exp. Pathol. 2012; 5: 367–73. 2. Widdel L, Kleinschmidt-DeMasters BK, Kindt G. Tumor-to-tumor metastasis from haematopoietic neoplasms to meningiomas: report of two patients with significant cerebral edema. World Neurosurg. 2010; 74: 165–71. 3. Honma K, Hara K, Sawai T. Tumour-to-tumour metastasis: a report of two unusual autopsy cases. Virchows. Archiv. Pathol. Anat. 1989; 416: 153–7. 4. Ventura DF, Vasquez KG. The incidental intrameningioma metastatic renal cells: first step in the diagnosis of systemic cancer. Rev. Chil. Neurocirugia 2013; 39: 180–1. 5. Caroli E, Salvati M, Giangaspero F et al. Intrameningioma metastasis as first clinical manifestation of occult primary breast carcinoma. Neurosurg. Rev. 2006; 29: 49–54. 6. Matyja E, Naganska E, Zabek M, Jagielski J. Meningioma with the unique coexistence of secretory and lipomatous components: a case report with immunohistochemical and ultrastructural study. Clin. Neuropathol. 2005; 24: 257–61. 7. Moller-Hartmann W, Herminghaus S, Krings T et al. Clinical application of proton magnetic resonance spectroscopy in the diagnosis of intracranial mass lesions. Neuroradiology 2002; 44: 371–81.

Elena How,* MBBS Richard Lee,† MBBS Kais Kasem,‡ MBBS Teresa Withers,* MBBS *Department of Neurosurgery, Gold Coast University Hospital, Southport, Queensland, Australia, †Department of Medicine, Greenslopes Private Hospital, Brisbane, Queensland, Australia, and ‡Department of Pathology, Gold Coast University Hospital, Southport, Queensland, Australia doi: 10.1111/ans.13045

© 2015 Royal Australasian College of Surgeons

Surprise within a meningioma: case report of signet ring cell carcinoma metastasis in a meningioma.

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