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Case Report
Revised

Case Report: Rare site for intraoral meningioma

[version 2; peer review: 2 approved, 1 approved with reservations]
PUBLISHED 06 Apr 2020
Author details Author details
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Abstract

Extracranial meningioma is very rare with few cases reported, especially in the oral cavity. Its diagnosis is considered a challenge owing to the unusual site of occurrence.  We report, to the best of our knowledge, the first case of extra-cranial meningioma as a primary tumor in the hard palate with no detected intracranial extension. A 59-year-old Egyptian female patient presented with a 22-year history of a large painless swelling at the right side of the hard palate, which could not be seen on radiographs.  An incisional biopsy was taken and, after assessment with a panel of immunohistochemical markers, the lesion was diagnosed as extracranical grade I mengiothelial meningioma. The patient did not show up for surgical excision and follow-up was not performed because of the lose of contact with the patient. Intraoral meningioma is a rare tumor. Immuohistochemical markers are important for confirming this diagnosis.

Keywords

Intra-oral meningioma, Benign tumor, Ectopic meningioma, Palatal lesion

Revised Amendments from Version 1

We have corrected grammar and language mistakes.
We have added a new reference and changed the numbering of citations and added the data to text.
We have added some lesions to the differential diagnosis list and discussed them.

See the authors' detailed response to the review by Eman Abdelzaher
See the authors' detailed response to the review by Maha M. Abdelsalam

Introduction

Meningioma is a benign neoplasm of meningothelial cells1. Meningioma may develop as a direct extension of a primary intra-cranial meningioma or as a true primary extra-cranial meningioma2.

Extra-cranial (ectopic) tumors are mostly seen in the head and neck region with no connection intra-cranially3. The most common extra-cranial site is the orbits. Meningioma arising in the oral cavity is extremely rare4. To the best of our knowledge, 19 cases have currently been reported in the oral cavity2,420 and we are reporting the first case in the hard palate.

Case report

A 59-year-old female patient presented to the outpatient clinic in the Oral and Maxillofacial Surgery Department, Cairo University in January 2019 complaining of a large painless swelling in the hard palate (Figure 1). The patient reported that the swelling had been present in her oral cavity for 22 years. The patient’s medical and familial histories were unremarkable. As well as there was not a history of exposure to radiation. Upon clinical examination on the day of admission, a large hard palatal swelling (3 cm × 3 cm) was evident on the right side of the hard palate. The swelling was covered by normal mucosa and showed a slight bluish tinge. A provisional diagnosis of a benign peripheral nerve neoplasm and a minor salivary gland benign neoplasm were made. CT scan was performed with no evidence of bone involvement.

1a1644ac-f447-4085-91f7-8c39deba3023_figure1.gif

Figure 1. Preoperative clinical picture showing 3 × 3cm swelling in the hard palate.

An incisional biopsy of the lesion was performed. Hematoxylin and eosin stained sections revealed meningothelial cells arranged in lobules. The cells exhibited round to oval nuclei (Figure 2). Psammoma bodies were also present (Figure 3). No mitotic activity and no cellular atypia were found. Immunohistochemical staining for tumor-associated markers was performed to confirm the diagnosis of meningioma and to exclude other mimic tumors as metastiatic carcinomas, schwannoma, neurofibroma, paraganglioma and perineurioma. Cells were positively stained using primary antibodies for epithelial membrane antigen (EMA) and vimentin (Figure 4a, b), but were not stained when using primary antibodies for S100, pancytokeratin, p63, chromogranin and renal cell carcinoma glycoprotein (Figure 5a–e).

1a1644ac-f447-4085-91f7-8c39deba3023_figure2.gif

Figure 2. Hematoxylin and eosin-stained sections showing epithelioid cells forming meningiothelial whorls (magnification, ×100).

Indistinct cell membranes with uniform nuclei and no mitotic figures (inset; magnification, ×200).

1a1644ac-f447-4085-91f7-8c39deba3023_figure3.gif

Figure 3. Hematoxylin and eosin-stained sections showing syncytial cells (magnification, ×100).

Psammoma bodies seen between meningiothelial cells (inset), (×200).

1a1644ac-f447-4085-91f7-8c39deba3023_figure4.gif

Figure 4.

Meningioma tumor cells showing a positive cytoplasmic immunohistochemical reaction for (a) epithelial membrane antigen and (b) Vimentin (magnification, ×200).

1a1644ac-f447-4085-91f7-8c39deba3023_figure5.gif

Figure 5.

Meningioma tumor cells react negatively following immunohistochemical staining for (a) renal cell carcinoma glycoprotein, (b) S100, (c) chromoginin, (d) p63, (e) PanCK (magnification, ×100).

No therapy was administered to the patient during her admission. Unfortunately, the patient did not show up for surgical excision and follow-up.

Table 1. Clinicopathological and radiographic data of the documented cases of extracranial meningioma.

StudyAge,
years
GenderSiteTumor sizeRadiographic
findings
TreatmentFollow-up
Brown et al.569MMaxillaNAML RLNot completed8 years
Simpson and Sneddon663FMaxillary alveolus4.5 × 2.7 × 2.7 cmWell-defined mixed
RL RO
Surgical excision.Under
review
Landini and Kitano748FMandibleNAWell-defined RLBlock resection2 years
Reddi et al.226FMaxilla3 cmIll-defined RLSurgical excision2 years
Kishore et al.844FSoft palate3 × 2 cmNSExcisional Biopsy4 years
Pfeifer et al.977FMaxilla (temporal
fossa )
NADense soft tissue
mass
Surgical resectionNS
Jones and Freedman1041FMandible4 × 2 cmWell defined RLExcisional biopsyNS
Jones and Freedman1074FMandible4 × 3 cmWell-defined RLExcisional biopsyNS
Kubotaa et al.1110MMandibleNAWell-defined RLEnucleated4 years
Mussak et al.1262MMandible7 × 3 cmWell-defined RLSegmental
mandibulectomy
NS
Lell et al.1340FMandibleNAWell-defined RLNSNS
Mosquede-Taylor et al.1453FMandible4 cmIll-defined mixed
RO RL
Surgical excision6 months
Rushing et al.15NAMandibleNA
Simsek and Komerik451FMaxilla2 × 2 cmIll-defined mixed
RL-RO
Surgical excision5 years
Pinting et al.1659MMaxillaNAWell-defined RLSurgical excision
and radiotherapy
NS
Maeng et al.1766FBuccal mucosa2 cmHeterogenously
enhanced mass
Surgical excisionYear and
half
Nair et al.1860FBuccal mucosa4 × 3 cmMass of
heterogeneous
density
Surgical resectionOne year
Rege et al.1935MMandibleNAIll-defined
ML RL
Partial resection5 years
Rommel et al.2020FMandible2 × 1.8 cmWell defined RLNo surgical
intervention.
One year

M, male; F, female; RL, radiolucent; RO, radioopaque; UL, unilocular; ML, multilocular; NA, not available; NS, not stated.

Discussion

Primary extra-cranial meningioma is an unusual tumor, especially in the oral cavity4. The first intraoral meningioma reported was by Brown et al. in 1976, which presented as a periapical radiolucency in the anterior maxillary region5.

To the best of our knowledge, 19 cases of primary meningioma in the oral cavity have been reported. Of these, 13 were in female patients, which is also true of the present case. However, the age range was wide in the reported cases – between 10 and 77 years old2,420; in the present case, the patient was 59 years old. Regarding the reported cases of intraoral primary meningioma, 6 of the 18 were in the maxilla2,46,9,16, 10 were in the mandible7,1015,19,20, 2 in the buccal mucosa17,18 and one in soft palate8. To the best of our knowledge, we report the first case in the hard palate.

The histopathological criteria of extracranial meningiomas are similar to those of their intracranial counterparts. All documented cases shared the same characteristics: whorls of spindle cells or epithelioid cell proliferation and psammoma bodies. In our case, diagnosis was challenging because of the tumor’s similarity with other tumor entities of peripheral nerve origin, as well as the uncommon location of the tumor. An immunohistochemical panel of tumor-associated markers was used to confirm the diagnosis and to avoid unnecessary aggressive treatment. Most of the 19 cases reported in the literature were diagnosis using immunohistochemical markers. All reported cases that used immunohistochemistry techniques to diagnose meningioma4,911,13,14,16,17,19,20 observed that the tumor cells stained positive for monoclonal antibodies against EMA and vimentin, with no immunoreactivity for S-100 protein, which was similar to our findings. However, EMA and vimentin are not useful to differentiate between meningioma and perineuroma as they both express positivity for EMA and vimentin but perineuroma the cells are spindle and elongated however, in our case they are rounded and polyhederal (meningiothelial pattern).

Unfortunately, our patient did not show up for surgical excision and follow-up was not done because of the loss of contact with the patient. However, most of the documented cases were treated successfully without recurrence by surgical excision. Some of the studies, such as that by Rommel et al.20, preferred only to follow-up with the patient rather than conduct surgical intervention. However, others preferred to perform aggressive treatment, such as as segmental mandibulectomy or segmental resection7,12

In conclusion, meningioma is a rare intraoral benign neoplasm. Immunohistochemical markers are an important tool to achieve a final diagnosis, especially for the differentiation from histological mimic entities of peripheral nerve origin, such as perineurioma and neurothekeoma and to avoid unnecessary aggressive treatment. Vimentin and EMA are the two important markers to confirm extra-cranial meningioma diagnosis.

Data availability

All data underlying the results are available as part of the article and no additional source data are required.

Consent

Written informed consent for publication of their clinical details and clinical images was obtained from the patient.

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Amer HW, Hafed L, Ibrahim S and Shaker S. Case Report: Rare site for intraoral meningioma [version 2; peer review: 2 approved, 1 approved with reservations]. F1000Research 2020, 9:95 (https://doi.org/10.12688/f1000research.21999.2)
NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article.
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ApprovedThe paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approvedFundamental flaws in the paper seriously undermine the findings and conclusions
Version 2
VERSION 2
PUBLISHED 06 Apr 2020
Revised
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Reviewer Report 18 Aug 2020
Hasan Onur Şimşek, Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Aydın Adnan Menderes University, Aydın, Turkey 
Approved
VIEWS 3
  • Ectopic Meningiomas are seen less frequently in oral regions, so it can be considered as a case report. However, the lack of follow-up of the case reduces the power of the article.
     
... Continue reading
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CITE
HOW TO CITE THIS REPORT
Şimşek HO. Reviewer Report For: Case Report: Rare site for intraoral meningioma [version 2; peer review: 2 approved, 1 approved with reservations]. F1000Research 2020, 9:95 (https://doi.org/10.5256/f1000research.25478.r68269)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
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Reviewer Report 06 Apr 2020
Eman Abdelzaher, Department of Pathology, Faculty of Medicine, Alexandria University, Alexandria, Egypt 
Approved
VIEWS 13
The requested ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Abdelzaher E. Reviewer Report For: Case Report: Rare site for intraoral meningioma [version 2; peer review: 2 approved, 1 approved with reservations]. F1000Research 2020, 9:95 (https://doi.org/10.5256/f1000research.25478.r62034)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
Version 1
VERSION 1
PUBLISHED 07 Feb 2020
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Reviewer Report 02 Mar 2020
Maha M. Abdelsalam, Department of Biomedical Dental Sciences, College of Dentistry, Imam Abdulrahman Bin Faisal University, Dammam, Saudi Arabia 
Approved with Reservations
VIEWS 13
Case Report: Case Report: Rare site for intraoral meningioma
 
The case report presents an extracranial meningioma of the hard palate. The patient’s gender is conformed to the tumor but there are (likely) no contributing or risk factors ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Abdelsalam MM. Reviewer Report For: Case Report: Rare site for intraoral meningioma [version 2; peer review: 2 approved, 1 approved with reservations]. F1000Research 2020, 9:95 (https://doi.org/10.5256/f1000research.24261.r59735)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response 06 Apr 2020
    Layla Hafed, Faculty of Dentistry, Ahram Canadian University, Cairo, Egypt
    06 Apr 2020
    Author Response
    Firstly, thank you doctor for your valuable comments. We added that data and answers that you commented to the new version of the manuscript. And I will answer them too ... Continue reading
COMMENTS ON THIS REPORT
  • Author Response 06 Apr 2020
    Layla Hafed, Faculty of Dentistry, Ahram Canadian University, Cairo, Egypt
    06 Apr 2020
    Author Response
    Firstly, thank you doctor for your valuable comments. We added that data and answers that you commented to the new version of the manuscript. And I will answer them too ... Continue reading
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16
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Reviewer Report 25 Feb 2020
Eman Abdelzaher, Department of Pathology, Faculty of Medicine, Alexandria University, Alexandria, Egypt 
Approved with Reservations
VIEWS 16
The case report is well written and presents an interesting case of ectopic meningioma. Some amendment is needed however.
  1. Radiological findings should be included. The authors merely mention that the lesion was not seen by radiology.
... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Abdelzaher E. Reviewer Report For: Case Report: Rare site for intraoral meningioma [version 2; peer review: 2 approved, 1 approved with reservations]. F1000Research 2020, 9:95 (https://doi.org/10.5256/f1000research.24261.r59777)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response 06 Apr 2020
    Layla Hafed, Faculty of Dentistry, Ahram Canadian University, Cairo, Egypt
    06 Apr 2020
    Author Response
    Thank you Dr. Eman for your valuable comments and for your time. We added that data and answers that you commented to the new version of the manuscript. And I ... Continue reading
COMMENTS ON THIS REPORT
  • Author Response 06 Apr 2020
    Layla Hafed, Faculty of Dentistry, Ahram Canadian University, Cairo, Egypt
    06 Apr 2020
    Author Response
    Thank you Dr. Eman for your valuable comments and for your time. We added that data and answers that you commented to the new version of the manuscript. And I ... Continue reading

Comments on this article Comments (0)

Version 2
VERSION 2 PUBLISHED 07 Feb 2020
Comment
Alongside their report, reviewers assign a status to the article:
Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions
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