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 Table of Contents  
ORIGINAL ARTICLE
Year : 2018  |  Volume : 35  |  Issue : 3  |  Page : 287-296

Orbitozygomatic approach results of 16 spheno-orbital hyperostotic meningiomas (en plaque)


1 Department of Neurological Surgery, Faculty of Medicine, Mansoura University Hospital, Mansoura, Egypt
2 Department of Pathology, Faculty of Medicine, Mansoura University Hospital, Mansoura, Egypt
3 Department of Radiology, Faculty of Medicine, Mansoura University Hospital, Mansoura, Egypt

Date of Submission10-Jun-2017
Date of Acceptance24-Aug-2017
Date of Web Publication07-Jan-2019

Correspondence Address:
Dr. Ashraf El Badry
Department of Neurological Surgery, Faculty of Medicine, Mansoura University Hospital, Mansoura, 35516
Egypt
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/bmfj.bmfj_119_17

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  Abstract 


Objective This study aimed to evaluate the results of the orbitozygomatic approaches of intraosseous meningiomas that causes hyperostoses involving the greater wing of the sphenoid and orbital bones with a small soft tissue component.
Patients and methods We carried out a prospective and retrospective study of 16 patients with meningiomas en plaque who underwent surgical procedures by the orbitozygomatic approach in the period between 1999 and 2014 at the Neurological Surgery Department, Mansoura University Hospital of Egypt.
Results The mean age of the patients was 49.6 years, whereas the most common complaint was proptosis accounting for 81.25% of cases. The second most common complaint was visual impairment accounting for 56.25% of cases. The orbitozygomatic approach was used for all the cases and drilling of the greater wing of the sphenoid, lateral orbital wall, orbital, and optic canal roof was performed. The small soft tissue component of the meningiomas was in the anterior and middle cranial fossa involving hyperostoses in all surrounding bones in all cases, whereas four cases showed intraorbital soft tissue meningioma. The results after surgery indicated an improvement of proptosis in 75% of cases, with less improvement in visual impairment observed in 56.25% of cases. Complications included third nerve palsy in 87.25% (14) of cases, and improved only in five cases. The extent of tumor surgical resection was subtotal in 75% (12) of cases. These tumors progressed in eight cases and only two patients underwent a second surgical intervention; others were referred to radiotherapy.
Conclusion Despite good surgical exposure in this approach, the radical excision of the tumors may be extremely difficult as the tumor in these areas showed involvement of very delicate structures (because of late medical consultation), which made the dissection impossible, but using this approach we can achieve good results including proptosis and visual impairment.

Keywords: hyperostoses, interaosseous meningioma, meningioma en plaque, skull base meningioma, spheno-orbital meningioma


How to cite this article:
El Badry A, Azeez AA, Khalik AA. Orbitozygomatic approach results of 16 spheno-orbital hyperostotic meningiomas (en plaque). Benha Med J 2018;35:287-96

How to cite this URL:
El Badry A, Azeez AA, Khalik AA. Orbitozygomatic approach results of 16 spheno-orbital hyperostotic meningiomas (en plaque). Benha Med J [serial online] 2018 [cited 2019 Oct 17];35:287-96. Available from: http://www.bmfj.eg.net/text.asp?2018/35/3/287/249408




  Introduction Top


The incidence of meningiomas is ∼15% of brain tumors and the majority of tumors are found in the sphenoid wing area [1]. Sphenoid ridge and calvarium meningiomas are commonly associated with hyperostoses in the surrounding bone that may have sheets of meningioma cells. Meningioma en plaque is defined as sheet or nodular-shaped tumors that produce adjacent hyperostoses [2]. The term was first reported by Cushing and Eisenhardt [3]. Shrivastava et al. [4] reported that this type of meningioma is present in 2% of all surgically treated meningiomas. However, Pompili et al. [5] reported a higher percentage, which is about 9% of all operated meningiomas. Most of the meningioma en plaque are present in the spheno-orbital area, but they may also appear in the convexity [6]. The differential diagnosis includes many lesions: fibrous dysplasia, optic nerve (meningioma, glioma, neuroma/neurofibroma), and inflammatory diseases of the optic nerve (multiple sclerosis optic neuritis, Paget’s disease, metastasis) [7].

Hyperostotic spheno-orbital meningioma may lead to proptosis, partial or total ophthalmoplegia, deterioration of vision, or may affect other cranial nerves, with a recurrence rate of about 20–30% in international series [8]. Computed tomography (CT) scan is a very good tool for visualization of the bone hyperostoses [6], whereas MRI (T2WE, T1WE postcontrast) is the best tool to show the tumor with its dural origin [9].

Cosmoses, decompression of the optic nerves, and other cranial nerves are the main purposes for such cases surgery. However, surgery in this territory may lead to severe complications as it is related to vulnerable cranial nerves, main skull base vessels, and cavernous sinuous [10], especially in recurrent cases with distorted anatomy.


  Patients and methods Top


We carried out a prospective and retrospective study of 16 patients with meningiomas en plaque (extensive full-thickness hyperostotic bony involvement) who underwent surgical procedures by the orbitozygomatic approach in the period between 1999 and 2014 at the Neurological Surgery Department, Mansoura University Hospital of Egypt. We performed thorough and complete recording of clinical and neurological examination data, presenting symptoms, and duration. All 16 patients were followed radiologically by CT and MRI to determine the extent of surgical resection and clinically every 6 months for the first year and once yearly thereafter.

The imaging investigations of CT were performed to define the hyperostotic part, whereas MRI was used to show the intradural soft fleshy part, cranial nerves and skull base vessels, veins, and sinuses.

Our findings indicated that the majority of patients were women [68.75%, 11 cases]; there were five male patients. The age of the patients ranged between 31 and 67 years; the mean age was 49.6 years. The most common complaint was proptosis in 81.25% (13 patients) of cases, whereas the second most common complaint was visual impairment in 56.25% of cases. Cosmetic disfiguring was present in all cases, but this was not the main complaint. The duration of symptoms at presentation varied from 6 months to 5 years (median: 13.2 months). Hyperostosis was present in all 16 patients (all located at the sphenoid ridge, the lateral and superior walls of the orbit in 62.25% of cases) ([Table 1]).
Table 1 Manifestation of 16 cases of meningioma en plaque

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Surgical technique

In operative theater we placed the patient in a supine position with the head fixed by pins, variable degree of rotation (30°–60°) opposite the approach side with increasing the rotation angle where the tumor was located more anteriorly and let the zygomatic arch be the uppermost part [11]. The curved or the question mark skin flap was performed beginning 1 cm anterior to the tragus to preserve the superficial temporal artery mainstem and anterior division intact. We also aimed to preserve the frontal branch of the facial nerve as it runs anteriorly, crossing the zygomatic arch. This skin incision must be posterior to the hairline for cosmetic reasons ([Figure 1]).
Figure 1 The patient’s position and the curved skin incision for the orbitozygomatic approach, which may reach the midpapillary contralateral side to provide adequate room to create an appropriate ipsilateral fronto-orbital bone flap by Zabramski et al. [12].

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Then, the scalp flap was elevated with the temporal fascia without aggressive dissection to the subgaleal fat pad to avoid injury to the fascial nerve branches ([Figure 2] and [Figure 3]) [12].
Figure 2 (a) The temporal fascia dissection and its elevation with the scalp flap; to avoid injury to the frontal facial nerve, we must leave a short cuff of the fascia at the insertion in the frontal bone for approximation during anatomical closure. (b) Part of the lateral and superior orbital rim and the zygoma arch before elevation of the temporal muscle [12].

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Figure 3 (a–d) Surgical technique of the orbitozygomatic approach [12].

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We then made the galea and the pericranium incision, followed by dissection in the direction of the superior orbital rim. We preserved the supraorbital neurovascular bundle by freeing it through the orbital rim. The periorbita was also freed entirely from the superior and lateral orbital walls using a sharp dissector.

The temporalis muscle was elevated and directed toward the posteroinferior corner of the skin flap. This technique was adopted in the surgery to maintain fascial nerve integrity. By this step, we exposed the entire target operative area, which included the sphenoid, the temporalis squama, and part of the frontal bone, and the zygomatic bone orbitozygomatic approach (frontotemporal with dislocation of the zygomatic arch) was used for all cases.

In our surgical procedure we used one segment bone flap technique (Mefty modified technique) by make the orbitozygomatic flap one segment (six bone cuts principle were applied begin by a cut through the root of the zygomatic process ([Figure 3]) [12] or splitting it in two bone flap a the orbitofrontal is the first segment contain the frontal part and orbital rim anterior part of orbital floor. The second segment contained the temporal and zygomatic part after splitting the zygomatic arch for more space and expanding the vision angle under a microscope. We had to protect the released periorbita with blade retractors. The superior and lateral walls of the orbit were cut by retracting the frontal and temporal dura using a high-speed saw blade. We used an operating microscope and microsurgical tools in all cases.

Drilling of the greater wing of the sphenoid extradural clinoidectomy, the lateral orbital wall, and the orbital and optic canal roof was performed in these cases using a high-speed drill including hyperostotic bone in the next phase.

We performed deroofing of the superior orbital fissure to visualize the periorbital fascia ([Figure 4]) [12].
Figure 4 (a, b) The cut through the lateral wall and roof of the orbit by a saw and provide large operative field with adequate angle to work by microscope [12].

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In the same way, we performed deroofing of the optic canal extradurally, but after identification of the optic nerve intradurally, especially if there was excessive hyperostoses around the optic canal, and made an incision in the dural cuff of the optic nerve for a short distance to release the compression by cutting the falciform ligament [12],[13]. The sylvian fissure was opened to approach the tumor from the superior surface. The small soft tissue component of the meningiomas was in the anterior and middle cranial fossa involving hyperostoses in all the surrounding bones in all cases, whereas four cases showed intraorbital soft tissue meningioma. Resection of the dura and the intradural portion of the tumor was performed. In five cases, the lateral wall of the cavernous sinus was partially affected and we did not perform excision. In four cases, periorbital extension was observed; thus, partial excision was performed. Dural repair was performed with a fascia lata graft with fibrin glue to achieve as watertight a closure as possible and the lateral walls of the orbit were reconstructed with titanium mesh and bone cement in some cases. We used lumbar drain in seven cases for a short period postoperatively. We obtained first biopsy from the involved bone then taking another sample from the soft tissue.

Case report

A 49-year-old woman presented with a complaint of headache 5 years ago, and after 3 years, she developed right eye proptosis. On examination, there was a noticeable elevation of the right anterior temporal and frontal bone elevating eyebrow. Visual acuity had deteriorated to 6/60 in the right eye and 6/9 in the left eye with the right partial third nerve. There was considerable hyperostoses of the right sphenoid bone beside the lateral wall and roof of the orbital, and frontal bone compressing the right optic nerve and superior orbital fissure; all these findings were obtained by CT brain axial cuts with bone window ([Figure 5] and [Figure 6]). MRI (T2WE, T1WE with gadolinium contrast) was performed and showed enhancement of the dura sheet as in the right sphenoid ridge and the right superior periorbital part without hyperostotic bone enhancement ([Figure 7]); most of the hyperostoses was removed using a high-speed drill including that in the lesser wing of the sphenoid bone, the lateral wall of the orbit and the superior. We decompressed the orbital fissure to relieve pressure on the third nerve. We opened the basal temporal dura and then we removed the carpet sheet-like meningioma tumor with its dural origin as much as possible. The periorbital part of the tumor was also excised as much as possible. A pericranium graft was used to close the deficient dura part, which was closed tightly by stitches and fibrin glue. Cranioplasty was performed by bone cement to cover the temporal bone and the lateral wall of the orbit defect ([Figure 8],[Figure 9],[Figure 10]). Histopathological investigation of the hyperostotic bone biopsy and sheet-like dural tumor showed clusters of meningioma cells ([Figure 11]). Postoperative CT brain axial cuts and bone window almost remove of the meningioma en plaque.
Figure 5 Three-dimensional computed tomography of skull of the patient showing proptosis and hyperostosis of the right frontotemporal region.

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Figure 6 Computed tomography brain and orbit axial cuts showing hyperostosis of the right greater, lesser wing of the sphenoid, lateral orbital wall.

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Figure 7 T2WE brain coronal cuts showing right periorbita extension of the tumor.

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Figure 8 Postoperative computed tomography of brain axial cut showing complete removal of meningioma en plaque.

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Figure 9 Postoperative computed tomography brain axial cut with a bone window showing complete removal of hyperostotic bone.

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Figure 10 T2WE brain postcontrast axial cuts postoperatively showing complete decompression of the left optic nerve.

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Figure 11 Histopathological investigation of the hyperostotic bone biopsy and sheet-like dural tumor showing clusters of meningioma cells.

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Another case of meningioma en plaque showed hyperostoses involving the right greater, lesser wing of the sphenoid, the orbital apex, and around the optic canal that appeared in CT brain axial cuts ([Figure 12] and [Figure 13]). MRI (T1WE with gadolinium contrast) showed enhancement of left meningioma en plaque as a small soft tissue entering into the inner sphenoid wing and extending through the orbital apex periorbital extension of the tumor ([Figure 14]). We used the orbitozygomatic approach and then all of the hyperostotic bone was removed using a high-speed drill. The optic nerve and the superior orbital fissure were decompressed and finally we removed the carpet sheet meningioma that entering into the inner greater wing of sphenoid and around the optic nerve with reconstruction of the dural defect by a pericranium graft. Then, watertight closure was performed by stitches and fibrin glue. Cranioplasty was performed by bone cement to cover the temporal bone and part of the lateral wall of the orbit defect. Postoperative CT brain axial cuts were performed ([Figure 15]). The pathology slides of hyperostotic bone and tumors proved the presence of atypical meningioma ([Figure 16]).
Figure 12 Computed tomography brain and orbit axial cuts showing hyperostosis of the right greater, lesser wing of the sphenoid, orbital apex.

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Figure 13 Computed tomography brain and orbit axial cuts showing hyperostosis of the left greater, lesser wing of the sphenoid, orbital apex and around the optic canal.

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Figure 14 T1WE brain postcontrast, axial cuts showing left meningioma en plaque small soft tissue entering into the inner sphenoid wing, and periorbita extension of the tumor.

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Figure 15 Postoperative computed tomography brain axial cut with a bone window showing complete removal of hyperostotic bone.

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Figure 16 Histopathology examination of meningioma en plaque showing high MIB with atypical meningioma WHO grade II. MIB-1 Labeling Index a cell proliferation marker.

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  Results Top


The results of histopathological examination showed meningiomas grade 1 in 13 cases (meningothelial six cases with Ki-67 proliferation index of average 1.7% and progesterone receptor presented in 56.25% of the cases, transitional type in two cases, mixed in three cases. Grade 2 (atypical meningioma) was found in three cases according to the WHO classification. The mean proliferation index was 5.2% Ki-67 and progesterone receptor positivity was present in only one case. The results after surgery showed an improved cosmetic appearance in all cases. The proptosis improved in 75% of cases, with less improvement in visual impairment only in 56.25% of cases, and seven cases showed no improvement, which may have been because of already poor vision preoperatively.

Complications in our series included :third nerve palsy in 87.25% of cases [14 cases: eight cases of partial third nerve palsy (two of them preoperative) and six cases of total third nerve palsy], and improved only in five cases (four cases of partial third nerve palsy and a cases of total third nerve palsy).

Six patients developed fascial numbness, which improved only in two cases. The extent of tumor surgical resection was subtotal in 75% of cases (12 cases: five cases with cavernous sinus extension, four with preorbital extension, and the rest with extensive involvement of the anterior and middle skull base). Subcutaneous collection appeared in four cases and was treated by acetazolamide and lumbar drain. One patient developed wound disruption with infected wound manifestation that was treated by surgical debridement and intravenous antibiotics for a couple of weeks.

These tumors progressed in eight cases; only two patients underwent a second surgical intervention. Others were referred to radiotherapy because of unfavorable surgical condition or because of the patient’s preference after consultation.


  Discussion Top


Meningioma with hyperostoses in the skull base (anterior and middle) is relatively common. It may also invade in variable degree the orbital walls and roof [14]. The mean tumor mass location determine tumor origin and its name which may be interaosseous meningioma or meningioma with hyperostoses according to the literature [15]. However, some studies describe these tumors as meningioma with hyperostoses for any case with dural involvement [16]. A recent classification for extradural meningiomas included purely extracalvarial (type I), purely calvarial (type II), or combined (type III) by Brotchi and Bonnal [17]. This classification may give the calvarium meningiomas (types II and III) other descriptive names: convexity (C) and skull base (B) [18]. Our study focused on spheno-orbital meningiomas associated with hyperostoses that had dural sheet involvement (en plaque) (types IIB and IIIB) or groups V and VI according to the Roser et al. [20] classification ([Figure 17]) as it is more specific in the definition of meningioma en plaque.
Figure 17 (a) Classification of globoid sphenoid wing meningioma that had osseous involvement. (b) Classification of en plaque carpet sheet-like sphenoid wing meningioma with hyperostoses. (c) Classification of purely bony sphenoid wing meningioma after Roser et al. [19].

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Many theories exist on the development of hyperostotic meningiomas, but the most popular theory is bone invasion by tumors [20]; this infiltration of the meningioma cells to the bone may be because of trauma, vascular impairment, enzymatic activity, or tumor cells of meningioma may abnormally activate osteoblasts, which leads to bone or tumor invasion into the bone [5]. The histopathological examination documents the presence of meningioma cells in the hyperostotic bone biopsies in all of our cases, which supports the tumor invasion theory. The extent of tumor resection is strongly related to improvements in neurological conditions, but is inversely related to recurrence and subsequent radiotherapy if needed [21],[22]. However, this may be very difficult because of presence of cranial nerves, cavernous sinus involvement, and skull base vessels, which may lead to severe postoperative disabilities as a complication of these surgeries, which exceed outweigh the recurrence sequelae [10].

Spheno-orbital meningiomas (en plaque) mostly result in symptoms and signs because of compression of a vital skull base structure. The main presenting symptom in our cases was unilateral swelling of the orbit (proptosis). The patients showed long duration of symptoms, with an average duration of 13.2 months, perhaps because of an insidious onset with little annoying symptoms made the patient reluctant to seek medical advice. All cases in our study showed cosmetic enhancement and the proptosis became better in 75% of cases postoperatively, especially with aggressive radical excision, on the basis of the short-term and long-term follow-up data obtained from other international studies [5].

An improvement in visual impairment occurred only in 56.25% of cases and seven cases showed no improvement, which may be because of already poor vision preoperatively.

Complications included third nerve palsy in 87.25% of cases (14 cases: eight cases of partial third nerve palsy and six cases of total third nerve palsy), which improved only in five cases (four cases of partial third nerve palsy and a case of total third nerve palsy). This high percentage of third nerve palsy could be related to many reasons including excessive hyperostotic bone at two lips of the superior orbital fissure, thermal or mechanical injury by a high-speed drill during the procedure of the approach (the step of making bone cut of superior and lateral orbital wall through the superior orbital fissure in bone flap formation), during superior orbital fissure decompression, vascular injury of perforators that feed third nerve, and surgical manipulation of the cavernous sinus part of the tumor. The most of improved was in cases who developed postoperative partial third nerve palsy that indicate minimal damage to the third nerve during the surgery for these cases. The onset of all cases of third nerve palsy postoperative except in two cases was present preoperative which may making the approach is responsible for this complication but I think this is related partially to the approach and mainly to the nature of this tumor which compress third nerve in cavernous sinus and in superior orbital fissure making decompression is very difficult whatever the approach especially this approach give a wide visual angle that help in surgical manipulation. Third nerve palsy reported by many researchers like Honeybul et al. (57%), Maroon et al. (no number was mentioned clearly), Sandalcioglu et al. (over 56.25% but permanent in two cases only) [23]. Perhaps, we had more patients with third nerve palsy, but this may be related to less experience at the beginning of the study, with improvements on the learning curve, the fact that patients presented at the late stage to seek medical advice, thermal injury by a high-speed drill or vibration, and may be the cause related to mechanical injury in the last step of removing the orbital part of orbitozygomatic approach bone flap (there is no evidence-based proof), may be also due to the change of anatomical landmarks including skull base foramen and fissures as a result of hyperostosis which may be handled by navigation system intraoperative (not present in our operative rooms).

Six patients developed fascial numbness, which improved only in two cases, perhaps because of heat injury from a high-speed drill that required continuous irrigation or injury during decompression. In particular, the results improved in few last cases because of cumulative experience from previous surgeries. Cushing and Eisenhardt [3] reported that visual impairment remained unchanged or improved in 63% of their patients. Pompili et al. [5] noted that visual manifestations became better in 48% of the patients and extraocular muscle palsy resolved or became better in 50% of the patients.

The degree of success of surgery for spheno-orbital meningiomas may be related to the degree of decompression of the optic nerve if the vision deterioration is related to optic nerve compression preoperatively. In our surgical technique, optic nerve decompression by optic nerve canal deroofing and anterior clinoidectomy was concept in doing surgery for these cases. The maximum hyperostotic bone removal must be achieved whenever possible to reduce recurrence as reported by Pieper and Al-Mefty [10], who even excised bone next to the hyperostotic parts to reduce the rate of recurrence. The absence of neuronavigation may be one of the factors that increase postoperative deficits − that is, permanent nerve palsy because of anatomy distortion by hyperostoses and cavernous sinus involvement that was reported in many papers [24].

These tumors progressed in eight cases; only two patients underwent a second surgical intervention. Others were referred to radiotherapy because of unfavorable surgical condition or because of the patient’s preference after consultation.

The data of our series goes with international studies that suggest subtotal excision of the tumor and limited removal of invaded bone because it was creeping in skull base and distorted anatomy by hyperostoses making complete removal very difficult.

We did not remove the cavernous sinus involvement as it has not been documented to yield better results with increased morbidity [25]. The invasion of the cavernous sinus plays the main role in recurrence [26]; many authors performed infiltration of the cavernous sinus and Zinn’s fibrous ring at the orbital apex for subtotal excision as it is the optimum approach to decrease morbidity [5]. The involvement of the Internal Carotid Artery (ICA) may be an indicator of the aggressiveness of the tumor and may not allow total excision of the meningioma [27].


  Conclusion Top


Despite good surgical exposure by this approach, radical excision of tumors may be extremely difficult as the tumors in these areas show involvement of very delicate structures (because of late medical consultation), making dissection impossible, but using this approach, we can achieve good results toward improvements in proptosis and visual impairment.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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    Figures

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