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Year : 2017  |  Volume : 60  |  Issue : 3  |  Page : 381-384
Meningioma with the unique coexistence of secretory and lipomatous components: A case report with immunohistochemical study


1 Department of Pathology, Grant Government Medical College, Mumbai, Maharashtra, India
2 Department of Pathology, B.K.L. Walawalkar Rural Medical College, Ratnagiri, Maharashtra, India

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Date of Web Publication22-Sep-2017
 

   Abstract 

Meningioma is the most common extra-axial neoplasm which accounts for 30% of all intracranial tumors with a female predilection. These tumors exhibit a broad spectrum of differentiation potency corresponding to different histological subtypes. Meningothelial cells can rarely show separate secretory or lipomatous (metaplastic) transformation. The coexistence of these two different histological subtypes in a single meningeal tumor is extremely rare with only two reported cases. We report an uncommon case of meningioma in a 40-year-old female showing histological admixture of both secretory and lipomatous (metaplastic) meningioma, which to our knowledge is only the third reported case in the world. It highlights the multipotency of phenotypic transformation of primary meningothelial cells. This rare tumor behaves in a fashion similar to benign meningioma of WHO Grade I type.

Keywords: Benign, lipomatous (metaplastic), meningioma secretory

How to cite this article:
Patil PR, Warpe BM, Juvekar VH, Manohar V. Meningioma with the unique coexistence of secretory and lipomatous components: A case report with immunohistochemical study. Indian J Pathol Microbiol 2017;60:381-4

How to cite this URL:
Patil PR, Warpe BM, Juvekar VH, Manohar V. Meningioma with the unique coexistence of secretory and lipomatous components: A case report with immunohistochemical study. Indian J Pathol Microbiol [serial online] 2017 [cited 2019 Dec 7];60:381-4. Available from: http://www.ijpmonline.org/text.asp?2017/60/3/381/215363



   Introduction Top


Meningiomas are slow-growing neoplasm of meningothelial cells attached at inner surface of dura mater, with remarkable morphological spectrum.[1],[2],[3] Their WHO classification grades them into three grades with 15 morphologic types of meningiomas [2] included in these grades.

The WHO Grade I meningiomas include two distinct variants of secretory and metaplastic meningiomas.[4]

Metaplastic meningiomas are characterized by focal or widespread mesenchymal components, which may be osseous, cartilaginous, myxoid, lipomatous, and xanthomatous, occurring singly or in combination.[2],[3] Among this list, lipomatous meningioma is reported by most authors as a meningothelial intracellular lipid accumulation rather than true metaplasia.[2],[3],[4],[5]

Secretory meningioma is characterized by the presence of eosinophilic hyaline inclusions defined as “pseudopsammoma bodies” which are periodic acid-Schiff (PAS) positive and not stained by von Kossa stain.[6]

Our meningioma case had combined features of lipomatous and secretory components, which is an infrequent histological subtype of benign WHO Grade I meningioma.[1],[5],[6] Till date, only two such previous cases of meningioma comprised secretory and lipomatous components have been reported,[5] thus making this case as only the third reported case in the world.


   Case Report Top


A 40-year-old Indian female presented with intermittent headache and progressive diminution of vision for the past 1 year. She denied any history of trauma or infective process and history was unremarkable. The physical examination revealed no palpable scalp mass or cranial nerve deficits. There was no abnormal gait. Sensory and motor examination was within normal limits. Systemic examination also did not reveal any abnormality.

With clinical diagnosis of intracranial space occupying lesion, the patient was subjected to magnetic resonance imaging (MRI) brain which revealed 6 cm × 4.4 cm × 4.1 cm, extra-axial dural-based tumor mass in the left frontoparietal region, compressing the brain with associated perilesional edema [Figure 1]a. The diagnosis of meningioma was offered and surgical excision was planned.
Figure 1: (a) Magnetic resonance imaging brain shows extra-axial, dural-based intra-cranial mass in the left frontoparietal region with perilesional edema. (b) Gross picture (left) shows a well-encapsulated globular meningeal mass. Cut surface (right) shows white, soft tumor with few cystic areas

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The tumor was completely resected and sent for histopathological examination. The gross examination of meningeal tumor revealed a well-encapsulated, globular mass of 5 cm × 5 cm. The cut surface was white, soft with few cystic areas [Figure 1]b.

On microscopy, in addition to typical whorls of meningothelial cells, the tumor showed two components, secretory areas with PAS-positive hyaline inclusions (pseudopsammoma bodies) and mature adipocyte-like cells [Figure 2]a, [Figure 2]b and [Figure 3]a. The adipocyte-like cells were positive with Oil Red O special stain [Figure 3]b.
Figure 2: (a) Microphotograph shows all three tumor components-meningothelial cells, adipocyte-like cells and secretory areas (H and E, ×40). (b) Microphotograph shows secretory component within the tumor (H and E, ×400). Inset - Pseudopsammoma bodies within the secretory areas (PAS, ×400)

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Figure 3: (a) Microphotograph shows adipocyte-like cells within the tumor (H and E, ×400). (b) Microphotograph demonstrates adipocyte-like cells within the tumor with special fat stain (Oil Red O, ×40). (c and d) Immunostaining procedure show strong positivity of tumor cells for Vimentin (left) and focal positivity for epithelial membrane antigen (right) of tumor cells

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The meningothelial cells were uniform tumor cells forming whorls surrounded by thin fibrocollagenous septae. Within whorls, epithelioid tumor cells were seen with fuzzy ill-defined borders, resembling a syncytium. These cells were polygonal with moderate amount of vacuolated eosinophilic cytoplasm, round to oval mildly pleomorphic nuclei, and inconspicuous nucleoli. There was no mitotic activity, necrosis, or hemorrhage. Also admixed with meningothelial cells, the tumoral lipid content resembled mature adipocytes with peripherally displaced nuclei [Figure 3]a and [Figure 3]b.

There were eosinophilic, PAS-positive pseudopsammoma bodies admixed with meningothelial cells. These bodies were loosely scattered or in groups, exhibiting eosinophilic spherical bodies surrounded by clear halo at places [Figure 2]b.

On immunohistochemistry (IHC), strong positivity was noted for Vimentin in both secretory and lipomatous areas [Figure 3]c. Tumor cells also showed focal positivity for epithelial membrane antigen (EMA) [Figure 3]d.

Based on histopathology and IHC, this rare case was reported as meningioma with coexistence of lipomatous and secretory components (WHO Grade I).


   Discussion Top


Meningiomas make up to 30% of all primary brain tumors,[4] which are slow-growing with many rare subtypes of meningioma.[3],[4] The incidence of this central nervous system tumor is higher in females with female-to-male ratio of 2:1 and it increases with age.[4],[7] Brain invasive (WHO Grade II), atypical (WHO Grade III), and rare anaplastic/malignant (WHO Grade III) meningiomas are rare and considerably more aggressive.[3],[4] Nearly 80% of all meningiomas are of the WHO Grade I type.[4]

Meningiomas are neoplasms arising from meningothelial cells, probably from pluripotent arachnoid cap cells.[4] Headache and seizures are common complaints, apart from complaints related to location, size of tumor, and evidence of raised intracranial pressure.[2],[4] Our patient had complaints of headache and diminution of vision without seizures.

On MRI, meningiomas are isointense to cerebral cortex and avidly contrast-enhancing extra-axial dural-based intracranial tumors. Areas of hemorrhage, cyst formation, or metaplastic alterations can be picked up well on radiological study.[2],[4] Furthermore, perilesional edema has been known to occur more commonly with secretory meningioma due to vascular endothelial growth factor production by tumor cells with resultant increase in blood supply to pial vessels.[2]

Due to complex embryogenesis of meninges, meningiomas present with many histological variants. The formation of mesenchymal tissue inside these tumors such as bone, cartilage, and fat is known to occur and is caused by metaplasia of the tumor cells. The subtypes of WHO Grade I meningioma incorporate metaplastic meningiomas.[7] The rare adipose form is one of the metaplastic differentiations of meningiomas called lipomatous/lipoblastic/vacuolated/lipo-meningiomas.[7] On extensive search only, around fifty reported cases of lipomatous meningiomas are found till date.[2] The detailed study of Mayo-clinic case series from1983–1998 revealed the incidence of lipomatous meningioma as 0.3%.[2]

It is recently called as lipidized meningioma over lipomatous meningioma because most authors agree that it does not represent a true metaplasia of meningothelial tumor cells. The later conclusion was based on ultrastructural study which revealed that lipid-laden cells featured desmosomes, interdigitating cell membranes and lipid droplets not bound to the cell membrane. This means that they occurred due to lipid accumulation which was secondary to metabolic abnormality of tumor cells rather than true metaplasia of tumor cells.[2],[5]

The cytoplasm of the lipidized tumor cells are filled with large fat droplets with nuclei shifted to the periphery and are mainly filled with triglycerides.[8] Tumoral lipid content varies from 10% to 30% with Oil Red O stain positivity.[1] The lipomatous variant must be differentiated from xanthomatous variant of meningioma. The later has tumor cells with centrally placed nuclei and numerous cytoplasmic fat vacuoles composed of cholesterol or glycogen.[8]

Lipomatous meningioma must be differentiated from lipoma and other malignant tumors such as metastatic mucinous carcinomas, giant-cell glioblastomas, pleomorphic xanthoastrocytomas, and some glioblastomas as treatment varies for them.[8]

Lipomatous meningiomas show both mesenchymal and epithelial properties as they show immunostaining for both Vimentin and EMA in the adipocyte-like tumor cells.[8]

Secretory meningioma is benign, WHO Grade I meningioma characterized by focal epithelial and secretory transformation of meningothelial cells. Its incidence reported in various studies ranged from 1.1% to 4.4% of all meningiomas, except one study which reported its incidence accounting for 9.3%.[6] Its female-to-male ratio is 9:1.[9]

This tumor is characterized by PAS-positive, diastase-resistant and von Kossa negative, “pseudopsammoma bodies.” These are small, round, eosinophilic bodies surrounded by clear halo at places.[6]

Secretory meningiomas have basic growth pattern of meningothelial type and rarely by mixed type or fibroblastic or angiomatous type.[6] The mechanism by which individual neoplastic cells containing intracytoplasmic lumina and accumulated secretory products on ultrastructural studies is unknown, but it is linked with altered glycosylation of tumor cells.[6] In addition, some pseudopsammoma bodies are seen to be located extracellularly, lacking obvious lumina on IHC.[6] On IHC, the secretory areas are positive for both epithelial, secretory markers (carcinoembryonic antigen, EMA, cytokeratin) and mesenchymal, Vimentin marker.[6]

EMA is most commonly used meningioma marker which yields patchy positivity in most meningiomas.[10] Vimentin is a useful marker to differentiate meningioma from metastatic carcinoma. Negative Vimentin immunoreactivity can exclude either a benign or malignant meningioma.[10] The assessment of proliferative index of tumor is measured with the antibody MIB-1, the clone of which targets the proliferation marker Ki-67 in paraffin embedded tissue. MIB-1 labeling indices above 5% suggest a greater likelihood of recurrence and is helpful as an adjunct to grading in borderline atypical meningioma cases.[10]

As strong positivity was noted for Vimentin and patchy positivity was noted for EMA in the tumor cells within adipocyte-like cells and in secretory areas, diagnosis of meningioma with histological admixture of both secretory and lipomatous (metaplastic) meningioma was made in our case.

Yamada et al.[11] diagnosed the first neurological case of secretory meningioma with lipomatous (metaplastic) component. Matyja et al.[12] reported the second case in a 58-year-old female with right temporal lobe tumor with similar diagnosis. In conclusion, this case is only the third reported case of secretory meningioma with lipomatous (metaplastic) component with dural-based, tumor mass in left frontoparietal region; it highlights the multipotency of phenotypic transformation of primary meningothelial cells. This rare tumor behaves in a fashion similar to benign meningioma of WHO Grade I type.

Acknowledgment

We would like to acknowledge the Department of Neurosurgery, Grant Government Medical College and Sir JJH, Mumbai, Maharashtra, India.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Jaiswal AK, Mehrotra A, Kumar B, Jaiswal S, Vij M, Behari S, et al. Lipomatous meningioma: A study of five cases with brief review of literature. Neurol India 2011;59:87-91.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Gasparinho MG, Ferreira M, Lavrador JP, Livraghi S. Revisiting lipomatous meningioma: A case report and review of an unusual entity. Int J Surg Pathol 2015;23:399-403.  Back to cited text no. 2
    
3.
Tang H, Sun H, Chen H, Gong Y, Mao Y, Xie Q, et al. Clinicopathological analysis of metaplastic meningioma: Report of 15 cases in Huashan Hospital. Chin J Cancer Res 2013;25:112-8.  Back to cited text no. 3
    
4.
Riemenschneider MJ, Perry A, Reifenberger G. Histological classification and molecular genetics of meningiomas. Lancet Neurol 2006;5:1045-54.  Back to cited text no. 4
    
5.
Kim L, Huang C, Morey AL, Winder MJ. Intraosseous lipomatous meningioma. Case Rep Neurol Med 2015;2015:482140.  Back to cited text no. 5
    
6.
Taraszewska A, Matyja E. Secretory meningiomas: Immunohistochemical pattern of lectin and ultrastructure of pseudopsammoma bodies. Folia Neuropathol 2014;52:141-50.  Back to cited text no. 6
    
7.
Carlotti Júnior CG, Colli BO, Chimelli L, Dos Santos AC, Elias Júnior J. Lipoblastic meningioma. Case report. Arq Neuropsiquiatr 1998;56:661-5.  Back to cited text no. 7
    
8.
Er U, Gürkanlar D, Kazancı A, Şimşek S, Bavbek M. Lipomatous meningioma: Report of a case and a diagnostic pitfall. Turk Neurosurg 2006;16:40-3.   Back to cited text no. 8
    
9.
Lakhtakia R, Ramdas GV, Alam A, Mehta A. Secretory meningioma mimicking malignancy. Med J Armed Forces India 2008;64:82-3.  Back to cited text no. 9
    
10.
Liu Y, Sturgis CD, Bunker M, Saad RS, Tung M, Raab SS, et al. Expression of cytokeratin by malignant meningiomas: Diagnostic pitfall of cytokeratin to separate malignant meningiomas from metastatic carcinoma. Mod Pathol 2004;17:1129-33.  Back to cited text no. 10
    
11.
Yamada H, Hanada T, Okuda S, Yokota A, Haratake J. Secretory meningioma with lipomatous component: Case report. Brain Tumor Pathol 1999;16:77-80.  Back to cited text no. 11
    
12.
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.  Back to cited text no. 12
    

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Correspondence Address:
Bhushan M Warpe
Department of Pathology, B.K.L. Walawalkar Rural Medical College, Ratnagiri, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_123_16

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