Surgical Management of Sphenoid Ridge Meningioma En Plaque (Spheno-Orbital Meningioma) Gasser Hasan Al-Shyal, Mohamed Soliman Mohamed, Mohammad Fathy Eissa

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Original article 295

Surgical management of sphenoid ridge meningioma en plaque (spheno-orbital meningioma) Gasser Hasan Al-Shyal, Mohamed Soliman Mohamed, Mohammad Fathy Eissa

Department of Neurosurgery, Faculty of Background and aim Medicine for Girls, Al-Azhar University, Cairo, The aim of the study was to investigate the surgical techniques to remove Egypt meningioma en plaque and to compare our technique and results with other Correspondence to Gasser H. Al-Shyal, MD, authors. PhD, FAANS, Department of Neurosurgery, Al- Patients and methods Azhar University Faculty of Medicine for Girls, Al-zahraa Hospital, Abbassya, Cairo, Egypt. This retrospective series case was done at our University Hospital. Eight patients Tel: +20-1006699718; were managed at Al-Zahraa University Hospital between March 2016 and March e-mail: [email protected] 2018. Preoperative and postoperative clinical and radiological assessments were Received: 14 January 2020 done for each patient. Revised: 11 May 2020 Results Accepted: 2 June 2020 There were eight patients in our study. All patients are women. The age range was Published: 30 October 2020 from 34 to 60 years and the mean was 45.4 years. Proptosis was the main clinical Al-Azhar Assiut Medical Journal 2020, manifestation and it was present in all eight patients. Three (37.5%) patients had 18:295–301 visual impairment while two (25%) of them had headache and retro-orbital pain. We achieved total resection in six (75%) out of our eight patients. All tumors were WHO grade 1 meningiomas. Regarding surgical outcome, proptosis improved in six (75%) of the eight cases. Visual impairment improved in two (66.7%) cases out of three who had visual declination preoperatively. Conclusion Meningioma en plaque can be safely removed without any morbidity or mortality. Proptosis and hyperostosis in the sphenoid bone are highly suggestive and this requires more accurate radiological studies. MRI with contrast is essential for these cases. Extensive bony drilling is required for gross total resection. It also facilitates resection of the soft part of the tumor with its dura. Some cases may need orbital wall reconstruction. Proptosis mostly improves after surgery.

Keywords: meningioma, proptosis, pterional approach, skull base, sphenoid wing meningioma, spheno- orbital meningioma

cavernous sinus, and temporal convexity. Hyperostosis Introduction may result in compression of the optic nerve and or Meningioma accounts for about one-third of the other cranial nerves. Also, it causes proptosis, visual primary brain tumors [1]. The term meningioma en deterioration, and cosmetic deformity [13,14]. plaque (MEP) was first described by Cushing [2], which is flat soft tumor that infiltrates the dura [3]. Proptosis is the most common presentation of MEP. It It invades the bone causing hyperostosis. This occurs is unilateral, nonpulsating, and irreducible. Other mostly along the sphenoid wing [4]. Two synonyms are manifestations include decreased visual acuity, used for the description of these tumors: MEP and headache, and limitation of extraocular muscle spheno-orbital meningiomas [5–8]. movements. Less frequently, swelling in the temporal region, seizure, and facial pain are seen [15]. These tumors are slowly growing, and it has either flat or slightly nodular shape [9]. MEPs are more likely to Differential diagnosis of these lesions is meningeal cause bony hyperostosis than are larger globular tumors sarcoidosis, osteoma, tuberculoma, and fibrous and the amount of hyperostosis is often dysplasia [16,17]. Choosing surgical approach and disproportionate to the relatively small tumor [10–12].

Hyperostosis may extend in the skull base involving the This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 lateral and superior orbital walls of the optic canal and License, which allows others to remix, tweak, and build upon the work the anterior clinoid process. The dural growth is usually non-commercially, as long as appropriate credit is given and the new extensive and may include the basal sphenoid wing, creations are licensed under the identical terms.

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strategy for removal of MEP are challenging, because it optic canal was opened to decompress the optic nerve has extensive orbital, bone, and dural involvement in cases with marked hyperostosis in the optic canal. If [18–21]. As a result, MEP has relatively high there was orbital infiltration, tumor in the orbit was recurrence rate than other types of meningioma [22]. followed and removed. We did not remove any tumor However, with the development in microsurgical part infiltrated superior orbital fissure dura to avoid technique and craniofacial approaches it is possible cranial nerve deficits. The blood supply MEP usually to be more radical in the management of these arises from branches of the middle meningeal artery. tumors and many surgeons reported favorable We got good devascularization of the tumor by outcomes [15,23–25]. In our opinion, extensive bony cauterizing these branches after resection of the drilling of the affected bone and good exposure of the sphenoid wing with hyperostotic bone. Soft tumors soft tumor mass is the key to achieving favorable are removed with the dura. The excised dura is replaced surgical outcomes. with the artificial substitute. We removed the affected bone to avoid recurrence and this leads to defects in lateral and/or roof of the orbit. We used a titanium Patients and methods mesh plate to replace drilled bones in the temporal fossa, This retrospective study was conducted at Al-Zahraa but we did not reconstruct the superior wall of the orbit. University Hospital, after approval from the local ethics All cases were examined in the same day after surgery committee and after written informed consent was and were followed up for 1 year. Follow-up imaging obtained from each patient before surgery. Eight (CT bone window and MRI) to access residual and patients were managed at Al-Zahraa University recurrent tumors was done after 6 months and 1 year. Hospital in the interval between March 2016 and March 2018. We reviewed the preoperative clinical Statistical analysis assessment for each patient. We did ophthalmology The study is an observational retrospective case series consultation for each patient to access their visual acuity. study and descriptive statistics was used as follows:

We did both MRI and computed tomography (CT) (1) Quantitative data: mean and SD were used to bone window (with 3D reconstruction) for every measure central tendency and dispersion. patient. We reviewed the preoperative images to (2) Qualitative data: the studied variables were access the location of the tumor, the site of analyzed using univariant analysis and presented hyperostosis, and to plan our surgical techniques. as frequencies and percentages (frequency distribution table) for qualitative variables. Axial and coronal CT scan images of the bone window were essential for visualization of hyperostosis. We found that contrast is essential for detecting the Results dural enhancing component of these lesions. In one There were eight patients in our study. All patients are of our cases, the lesion did not appear except in women. The age range was from 34 to 60 with a mean contrasted MRI. Large axial orbital cuts increase the age of 45.4 years (Table 1). sensitivity of picking up small dural enhancement. Clinical manifestations We did pterional craniotomy for all eight cases. Care Proptosis was the main clinical manifestation and it was taken to avoid injury of the frontal branch of the was present in all eight patients. One patient had facial nerve. In some patients, we removed the soft bilateral exophthalmos due to toxic goiter and the tumor that infiltrated the temporalis. After removal of tumor was accidently discovered after doing MRI on the extracranial component of the tumor and the orbits in the Ophthalmology Department. Three separation of temporalis, a high-speed drill was used (37.5%) patients had visual impairment while for removal of hyperostotic bone of the lesser and two (25%) of them had headache and retro-orbital greater sphenoid wings. In two cases, we flattened pain. the marginal tubercle (elevation of the posterior border of the zygomatic process of the frontal bone) Resection, pathological grading, and reconstruction to get good exposure of the lateral wall of the orbit and Gross total resection was done for six (75%) out of the anteroinferior part of the greater sphenoid wing eight patients (Fig. 2). In one of the other two patients, (Fig. 1). Then, the remaining part of the bone is we left the part invading the dura of the superior orbital elevated. We continued drilling of the pathological fissure and the residue did not increase in size within bone of sphenoid wings and roof of the orbit. The the follow-up period. In another one, we left a piece of [Downloaded free from http://www.azmj.eg.net on Monday, December 7, 2020, IP: 41.42.169.215]

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Figure 1

Increasing exposure with marginal tubercle drilling. Arrow points to the marginal tubercle: (a) before drilling and (b) after drilling.

Table 1 Summary of patients with meningioma en plaque Cases Age Sex Preoperative Grade of Tumor Bone Surgical results Complications clinical picture resection grade reconstruction WHO 1 55 F Proptosis, Total 1 Yes Headache and visual Subgaleal CSF collection headache, and impairment improved for 2 months, 3rd nerve visual impairment proptosis is the same palsy 2 45 F Proptosis Total 1 No Proptosis improved No 3 48 F Proptosis Total 1 Yes Proptosis improved No 4 50 F Proptosis, visual Total 1 No Proptosis improved, visual Frontal branch of VII n. impairment impairment improved injury, CSF collection for 1 month 5 34 F Proptosis, slight Subtotal 1 No Proptosis is the same, Severe declination of visual impairment severe declination in visual visual acuity acuity 6 30 F Proptosis, retro- Subtotal 1 Yes Proptosis improved No orbital headache 7 41 F Proptosis Total 1 No Proptosis improved No 8 60 F Proptosis Total 1 No Proptosis improved No CSF, cerebrospinal fluid; F, female.

the tumor that was closely adherent to the optic nerve. complications. One patient suffered from severe It showed increasing size during the follow-up. There declination of visual acuity due to traction of a piece was no recurrence for all cases after total excision of the of tumor that was very adherent to the optic nerve. Two tumor. All tumors were WHO grade 1 meningiomas. of our patients experienced cerebrospinal fluid leak and In three (37.5%) cases, we used titanium mesh to subgaleal collection that has been improved and reconstruct the lateral wall of the orbit and the disappeared after 2 months. One patient has third temporal fossa (Fig. 3). nerve palsy while another one has injury of the frontal branch of the facial nerve. Improvement in preoperative clinical manifestations Regarding surgical outcome, proptosis improved in six (75%) out of our eight cases. Visual impairment Discussion improved in two (66.7%) cases out of three who had MEP accounts for about 2–4% of intracranial visual declination preoperatively. meningiomas [8]. These tumors cause significant hyperostosis and a widespread, carpet-like, soft- Complications tissue growth at the dura. Commonly, it involves the Postoperative and follow-up periods were uneventful sphenoid ridge with the orbit, so it is also called for five patients. Three patients have some spheno-orbital meningioma [4]. [Downloaded free from http://www.azmj.eg.net on Monday, December 7, 2020, IP: 41.42.169.215]

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Figure 2

Preoperative and postoperative radiological images of left sphenoid wing MEP: (a) preoperative MRI axial T1 images showing dural enhancement of the sphenoid wing, (b) postoperative axial images showing removal of the enhancing lesion in the middle cranial fossa, (c) postoperative axial CT bone window showing removal of hyperostotic bone without reconstruction. CT, computed tomography; MEP, meningioma en plaque.

Figure 3

(a) Preoperative MRI of right MEP, (b) postoperative MRI after excision of the enhancing mass and drilling of the hyperostotic bone, (c) preopertaive CT scan showing hyperostosis in the sphenoid bone with red arrow pointing to the marginal tubercle, (d) postoperative CT scan bone window showing reconstruction of the temporal bone with titanium mesh. CT, computed tomography; MEP, meningioma en plaque. [Downloaded free from http://www.azmj.eg.net on Monday, December 7, 2020, IP: 41.42.169.215]

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Some authors in the past [9] have recommended not to cases. We found that drilling of the marginal tubercle do surgical intervention for these tumors be considered without orbitozygomatic craniotomy is enough to only as a last resort because of the high surgical improve angles of exposure and getting direct access mortality rate. Over the years, this concept has been to the hyperostotic bone in the lower sphenoid and changed so MEP can be resected safely without or with middle cranial fossa. Oya et al. [13] stated that minimum morbidity [23,26,27]. orbitozygomatic approach may cause enophthalmos because it separates the periorbita from the orbital In all studies we reviewed, most of the patients were rim. They stated that reconstruction of the orbit is women [4–7,20]. In a series done by Jesus and Toledo not necessary if the periorbita was not detached from [8], all patients were women. We found the same the orbital rim. regarding sex as all our patients are women. Li et al. [4] found that the mean age of MEP was 45.5 years. Oya et al. [13] reported gross total resection in 38.5% of This is what we found in our series. The mean age was their patients with MEP, while Jesus and Toledo [8] 48.3 years in a series of 37 patients studied by Oya et al. achieved total resection in 83.3% of cases. Schick et al. [13]. [29] did total resection for 59.7% of their MEP cases. We are in the same range as we achieved total resection Li et al. [4] found that all cases in their series presented in 75% of our cases. Terrier et al. [22] did total with proptosis and this is like what we found in our resection in 40% of their MEP cases. Furthermore, results. Oya et al. [13] found that five of his 39 patients they stated that their series did not show any significant did not have proptosis. difference in recurrence rate between total and partial tumor removal. We do not agree with this opinion as Visual impairment was present in two (33.3%) cases one of our cases with soft tumor residues showed an out of six in a series done by Jesus and Toledo [8]. increase in the size after 6 months. Honeybul et al. [20] reported the same results. They found that five (33.5%) of their 15 patients had visual Li et al. [4] found that 89% of their cases were of WHO deterioration. Our results are not far from this, as three grade 1 meningioma. In a study by Sandalcioglu et al. (37.5%) of our patients had visual impairment. Oya [28], they confirmed that all his cases were WHO et al. [13] found that preoperative visual deficits were grade 1. Histopathological examination of all present in 53.8% of their patients. specimens in our study revealed grade 1 meningiomas.

Li et al. [4] reported that 24.3% of the studied MEP Jesus and Toledo [8] used methyl acrylate to reconstruct patients had headache. Similar to that retro-orbital pain the defect in the temporal bone after removal of the and headache were present in two (25%) out of eight hyperostotic bone. They mentioned that the sphenoid cases in this series. Jesus and Toledo [8] found that one ridge does not need reconstruction. We used titanium case out of their six cases had headache. Honeybul et al. mesh to cover the bony defect in the temporal fossa and [20] found that 46.7% of their patients had headache. lateral wall of the orbit. Construction was done for 37.5% Jesus and Toledo [8] found that out of six MEP cases, of our cases without any difference in cosmetic outcome. one case presented with seizures. Opinion varies regarding reconstruction of the superior and thelateralorbital wall.Maroonetal. [18]reported no Different surgical approaches have been reported for cosmetic problems after performing 200 cases without excision of MEP including pterional, frontotemporal- reconstruction. Oya et al. [13] did not reconstruct any orbitozygomatic and cranio-orbital approaches orbital walls even after complete drilling of the roof and [5–8,18]. Jesus and Toledo [8] used frontotemporal posterolateral walls of the orbit. Gaillard et al. [24] craniectomy with orbital decompression for all their recommend orbital reconstruction if more than one cases. Terrier et al. [22] used frontotemporal approach orbital wall had been resected. We confirmed this in with extradural removal of the invaded or hyperostotic our study. Both superior and lateral walls were removed bone with high-speed drills and rongeurs. Oya et al. completely (till orbital apex) in one of our cases. [13] reported their use of the modified Dolenc Reconstruction was not enough to cover the lateral approach with extradural bone drilling. In our series, wall of the orbit. Proptosis have not been improved as all cases were done via pterional craniotomy with a result of protrusion of the temporal tip dura in the orbit. extradural bone drilling. Proptosis represents the main clinical symptom of Sandalcioglu et al. [28] did osteotomy of both upper MEP. Jesus and Toledo [8] reported proptosis and lateral orbital ridges to excise 81.2% of their MEP improvement in 100% of their patients. Honeybul [Downloaded free from http://www.azmj.eg.net on Monday, December 7, 2020, IP: 41.42.169.215]

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et al. [20] reported 84.6% improvement in their MEP (11.9%) of their 67 patients had transient patients. Oya et al. [13] documented that proptosis cerebrospinal fluid leak and were successfully treated improved in 73.5% of their cases. We are in the same by lumbar drainage (five patients) or puncture (three range as proptosis has improved in 75.5% of our patients). In the series by Honeybul et al. [20], they patients. document postoperative hemiplegia, DVT, pulmonary embolism, chronic subdural hematoma, and retro- Very important goal for surgery is to improve visual orbital hematoma with 6.6% incidence for each. acuity. Scarone et al. [30] document that visual None of our patients experienced any of these symptoms improved in 85% of their patients. complications. Cannon et al. [14] reported 16.6% improvement. et al. Oya [13] achieved 66.7% improvement of Conclusion visual acuity in their MEP patients and we got MEP can be safely removed without any morbidity or similar result as visual symptoms improved in two of mortality. Proptosis and hyperostosis in the sphenoid our three (66.7%) patients with preoperative visual bone are highly suggestive and this requires more impairment. accurate radiological studies. MRI with contrast is essential for these cases. Extensive bony drilling is Visual deterioration has been reported in many required for gross total resection. It also facilitates literatures. In the study by Honeybul et al. [20] resection of the soft part of the tumor with its dura. 26.6% of cases experienced visual deterioration after Some cases may need orbital wall reconstruction. surgery. Oya et al. [13] reported no (0%) postoperative Proptosis mostly improves after surgery. visual worsening in their MEP cases. In our series, one (12.5%) patient experienced severe visual deterioration. Financial support and sponsorship Regarding recurrence, our cases were followed up for Nil. 24 months (2 years) except for two cases that have been followed up for 21 months. Schick et al. [29] reported Conflicts of interest 10.4% within the 24-month follow-up period. In our There are no conflicts of interest. series, there was no recurrence in the six cases that have been excised totally. One of our cases that has been done subtotally showed an increase in tumor size after 1 References 1 Fogh SE, Johnson DR, Barker FG, Brastianos PK, Clarke JL, Kaufmann TJ, year. et al. Case-based review: meningioma. Neurooncol Pract 2016; 3:120–134. 2 Cushing H. The cranial hyperostosis produced by meningeal Complications may occur after MEP surgery. The endotheliomas. Arch Neurol Psychiatry 1922; 8:139–154. 3 Tsutsumi S, Izumi H, Yasumoto Y, Ito M. Convexity en plaque meningioma most common complications that has been reported manifesting as subcutaneous mass: case report. 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