- Department of Neurosurgery, Hospital Italiano de Buenos Aires, Ciudad Autonoma de Buenos Aires, Argentina
- Department of Pathological Anatomy, Hospital Italiano de Buenos Aires, Ciudad Autonoma de Buenos Aires, Argentina
- Department of Head and Neck Surgery, Hospital Italiano de Buenos Aires, Ciudad Autonoma de Buenos Aires, Argentina
- Reconstructive Surgery, Hospital Italiano de Buenos Aires, Ciudad Autonoma de Buenos Aires, Argentina.
Correspondence Address:
Emily Zoraida Guerra Davila, Department of Neurosurgery, Hospital Italiano de Buenos Aires, Ciudad Autonoma de Buenos Aires, Argentina.
DOI:10.25259/SNI_222_2023
Copyright: © 2023 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.How to cite this article: Pablo Ajler1, Emily Zoraida Guerra Davila1, Pedro Plou1, Florencia Casto1, Silvia Christiansen2, Luis Alejandro Boccalatte3, Juan Larrañaga4. Multidisciplinary approach to anaplastic and metastatic meningioma: A case report and review of the literature. 07-Jul-2023;14:230
How to cite this URL: Pablo Ajler1, Emily Zoraida Guerra Davila1, Pedro Plou1, Florencia Casto1, Silvia Christiansen2, Luis Alejandro Boccalatte3, Juan Larrañaga4. Multidisciplinary approach to anaplastic and metastatic meningioma: A case report and review of the literature. 07-Jul-2023;14:230. Available from: https://surgicalneurologyint.com/surgicalint-articles/12401/
Abstract
Background: Meningiomas are slow-growing neoplasms, accounting for 20% of all primary intracranial neoplasms and 25% of all intraspinal tumors. Atypical and anaplastic meningiomas are infrequent, representing fewer than 5% of all meningiomas. Unusually, they can show aggressive behavior, and extracranial metastases are extremely rare, representing approximately 0.1% of all reported cases.
Case Description: Fifty-six-year-old male patient diagnosed with atypical basal frontal meningioma with multiple resections, both endoscopic endonasal and transcranial. After hypofractionated radiosurgery, the patient showed new tumor recurrence associated to right cervical level II ganglionic metastasis. We opted for complete resection of the meningioma and reconstruction with anterior rectus abdominis muscle flap, as well as selective cervical ganglionectomy. Anatomical pathology showed neoplastic proliferation of meningothelial cells in syncytial cytoplasm, oval or spherical nuclei with slight anisocariosis and hyperchromasia, and intranuclear vacuoles, all compatible with anaplastic meningioma.
Conclusion: Due to a lack of consensus on how to treat a metastatic malignant meningioma, this pathology requires a multidisciplinary approach, and treatment needs to be adapted to each particular case. Complete resection of the lesion is the primary goal, and this requires complex procedures involving endocranial as well as extracranial surgeries, which result in composite defects difficult to resolve. Microvascular free flaps are considered the gold standard in reconstructions of large skull base defects, with high success rates and few complications.
Keywords: Anaplastic meningioma, Anterior rectus abdominis muscle flap, Deep inferior epigastric artery perforator flap, Metastatic meningioma, Skull base reconstruction
INTRODUCTION
Meningiomas are slow-growing tumors derived from the arachnoid “cap cells,” accounting for 20% of all primary intracranial neoplasms and 25% of all intraspinal tumors.[
Extracranial metastases of meningiomas are rare, accounting for 0.1% of all meningiomas.[
Despite advances in radiology and radiosurgery, the standard treatment of meningiomas is still surgical resection.[
We present the case of a patient with recurrent invasive anaplastic meningioma and cervical metastasis, its surgical resection, and later reconstruction. Biological aggressiveness, the unusual presentation, the surgical challenge, and the skull base reconstruction make this case valuable. A review of the literature available on this topic was also performed.
CASE DESCRIPTION
Fifty-six-year-old male patient, with a history of ChagasMazza disease, diagnosed in 1996 with anterior skull base meningioma due to headaches without any other accompanying symptoms. He underwent surgery in a different center before reaching our institution in 2016, with a recurrent meningioma with intranasal and right orbit invasion [
Figure 1:
Preoperative (a and b) and postoperative (c and d) gadolinium-enhanced T1-weighted magnetic resonance imaging of the first surgery (in 2016) in our institution. (a and b) Voluminous polylobulated expansive brain tumor with skull base compromise, orbital and nasal invasion. (c and d) Imaging control of gross total resection of the tumor.
Figure 2:
Anatomical pathology images of the first surgery in 2016 (a-c). (a) Focal positivity for progesterone receptor (Immunohistochemistry, ×40). (b) Increase in cellular kinetics (Ki 67 immunohistochemistry. ×40). (c) Cells with syncytium formation and conspicuous nucleoli. Mitosis figures are observed (Hematoxylin and eosin stain, ×40).
Figure 3:
Gadolinium-enhanced T1-weighted magnetic resonance imaging of the tumor recurrence and the neck metastasis in 2018. (a and b) A more extensive component of the expansive nasoethmoidal and orbital wall lesion can be observed. The lesion compromises the subcutaneous tissue of the nose. (c and d) Right laterocervical adenopathy compatible with metastasis (white arrow).
Figure 5:
Anatomical pathology images of the second surgery in 2018 (a-e). (a) Syncytial arrangement, moderate nuclear pleomorphism (H&E, ×40). (b) Nuclear pleomorphism, evident nucleoli and marked vascularization (H&E, ×40). (c) Areas of tumor necrosis and nuclear pleomorphism with some pyknotic nuclei (H&E, ×40). (d) Increase of cellular kinetics with “hot spots” of 12% with Ki67 (Immunohistochemistry, x40). (e) Neoplastic infiltration in lymph node. Note the presence of intranuclear vacuoles in the center of the field (H&E, ×40).
DISCUSSION
Meningiomas grades II and III are characterized by a more aggressive behavior and a high recurrence rate, ranging from 29% to 52% and 50% to 94%, respectively.[
Metastatic meningioma is a rare clinical entity, with an estimated prevalence ranging from 0.1% to 0.76% of all patients with meningiomas according to different reported series.[
In our case, after several surgical resections, the patient presented with recurrence at the level of the anterior cranial fossa with invasion of the superior meatus of the nasal cavity, medial orbital wall, and nasal subcutaneous tissue. Although the hematogenous route is the best known for metastatic meningiomas, our patient presented with lymphatic spread, since histopathologic findings confirmed that a cervical lymph node was infiltrated by neoplastic cells, compatible with metastasis.
There is no standard protocol to treat metastatic meningiomas.[
Due to tumor aggressiveness, high cellularity, and presence of metastasis, we opted for total resection with negative surgical margins, later reconstruction with a microvascular free flap, and selective ipsilateral metastatic cervical lymph node drainage. Even though most reconstructions for large skull base defects are secondary to resections of locally aggressive malignant lesions, surgical resolution of locally aggressive benign tumors may also cause large defects that must be repaired.[
Literature frequently mentions the free rectus abdominis muscle flap as the most widely used in microvascular free tissue transfer reconstruction of the skull base, due to its reliable anatomy and vascularity, its irrigation by the inferior epigastric artery, and a branch of the external iliac artery.[
CONCLUSION
Due to a lack of consensus on how to treat metastases of a malignant meningioma, we believe that this pathology must have a multidisciplinary approach, and treatment needs to be adapted to each particular case. Total resection is the primary goal for the treatment of this disease since adjuvant therapies are not highly beneficial. Microvascular free flaps are considered the gold standard in large skull base reconstructions, with high success rates and minor complications.
Declaration of patient consent
Patient’s consent not required as patient’s identity is not disclosed or compromised
Financial support and sponsorship
Publication of this article was made possible by the James I. and Carolyn R. Ausman Educational Foundation.
Conflicts of interest
There are no conflicts of interest.
Disclaimer
The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.
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RAMIRO DEL VALLE - ROBLES
Posted July 20, 2023, 12:31 am
I suggest radio-theranostic option as a supplemental treatment with lutetium
( 177Lu-DOTATOC), in recurrent anaplastic and atypical Meningiomas with SSTR2 target expression.
Surgery and Radiosurgery are focal therapy, on the other hand, Theranostic is personalized precision therapy beyond the tumor limits : Nanomedicine