| Home | E-Submission | Sitemap | Contact Us |  
top_img
The Nerve > Volume 10(2); 2024 > Article
Kim, Woo, and Kim: Spinal Upper Thoracic Extradural Meningioma: A Case Report and Literature Review

Abstract

The most common extradural spinal lesions are metastatic tumors. However, approximately 2.5% to 3.5% of spinal meningiomas are extradural meningiomas. This article describes a rare case of a thoracic extradural meningioma. A 58-year-old woman was admitted following two months of gait disturbance. Magnetic resonance imaging suggested an extradural meningioma at the T1-5 level. Radical surgical resection was performed, and the lesion was pathologically identified as meningothelial meningioma. Additionally, we reviewed and summarized 12 cases of spinal extradural masses since 2018. Based on the literature, we discuss the location, pathology, and clinical outcomes of these uncommon lesions.

INTRODUCTION

Spinal tumors are to be classified as extradural, intradural-extramedullary or intramedullary. The most prevalent intradural lesions comprise schwannomas, neurofibromas, and meningiomas, while the most common metastatic lesions occur in the epidural space14).
Spinal meningiomas are typically considered primary spinal tumors, accounting for up to 20% to 30% of all primary spinal tumors3). These tumors are predominantly located in the intradural and extramedullary compartments of the thoracic region13); however, extradural meningiomas are extremely rare2,26). Spinal extradural meningiomas account for only 2.5% to 3.5% of all spinal meningiomas15,21,25). In this report, we present a case of an extradural meningioma in the upper thoracic spine and review existing literature on epidural meningiomas.

CASE REPORT

A 58-year-old female presented with progressive motor weakness and paresthesia in both legs over the course of two months. She exhibited paraparesis with a motor grade of 4 in both legs and experienced gait disturbances. Nonetheless, she reported no symptoms of sphincter dysfunction.
A spinal magnetic resonance imaging (MRI) of the thoracic vertebrae revealed an 8.8 cm mass in the left and posterior extradural space at the T1-5 level, showing high signal intensity and homogeneous, avid enhancement on T1 and T2 images. The mass compressed the thecal sac and displaced the spinal cord to the right, causing spinal cord edema at levels T1-3 (Fig. 1). A subsequent computed tomography scan of the thoracic spine identified calcification on the right side of the mass without any evidence of bony erosion (Fig. 2). Preoperatively, endovascular embolization was performed on the feeding artery of the mass, which originated from the right subclavian artery, to reduce the risk of bleeding during surgery. The following day, the patient underwent excision involving a total laminectomy at T2 and T3 and a partial hemilaminectomy at T1 and T4. The laminectomy revealed a reddish-yellow tumor compressing the spinal cord (Fig. 3). A biopsy of the T1-4 epidural lesion identified a meningothelial meningioma, classified as World Health Organization (WHO) grade 1, showing mild cellular atypia and mitotic activity (Fig. 4). Post-surgery, the patient did not receive additional treatments such as postoperative radiotherapy and was discharged on the 12th day after the surgery. She was advised to return for follow-up visits in the 1st, 2nd, 3rd, and 5th months postoperatively. One month later, a follow-up spinal MRI revealed a residual enhancing lesion at the left neural foramina of T2-3 and T3-4 levels, as well as enhancement at the posterior epidural space and laminectomy site at T3-5. Additionally, we observed postoperative fluid collection in the soft tissue from T1-5. Despite these findings, the patient's neurological symptoms had improved, and she returned to work without further complaints (Fig. 5).
Using an operative microscope, the patient underwent a radical surgical excision of the lesions through a total laminectomy at T2 and T3, and a left partial hemilaminectomy at T1 and T4. An extradural tumor was discovered intraoperatively. The lesion, characterized by its reddish-yellow color, hard consistency, and calcifications, had severely compressed the spinal cord to the right. Intra-operative monitoring was used, and there were no special abnormalities during surgery. The extradural mass was completely removed.
A study reported that lamia replacement using a titanium plate improved spinal stability18). In this surgery, lamina replacement was not performed and no instability or dead space occurred. However, if there is a possibility of instability during the surgical procedure, posterolateral fusion or laminoplasty can be considered. In order to reduce dead space, in this surgery, suture was performed densely, as if sutured the fascia, rather than approximately sutured the muscle.

DISCUSSION

Spinal meningiomas primarily occur within the dura of the thoracic spine. These tumors are believed to originate from the dentate ligament, with over 95% pathologically classified as WHO grade I19). Although pure epidural meningiomas are rare, it is crucial to differentiate these extradural lesions from metastatic tumors preoperatively and intraoperatively, as they can be easily misidentified19). Extradural spinal meningiomas typically exhibit the same histologic features, are most frequently diagnosed in individuals in their 50s and 60s, predominantly occur in the thoracic spine, and are more common in females compared to intracranial meningiomas15).
Clinically, the symptoms of extradural meningiomas resemble those of intradural meningiomas, including back pain, sensory and motor changes, and, in some cases, sphincter dysfunction1).
Surgical resection of extradural meningiomas can be challenging and often necessitates a second surgical approach11,13). In our case, the patient underwent a T2-3 total laminectomy and a left partial hemilaminectomy at T1 and T4. The tumor, which had severely compressed the spinal cord, was nearly completely resected. To prevent cord injury, a safe surgical field where surgical instruments can enter and exit was secured through a total laminectomy. Additionally, a cottonoid was inserted between the capsule membrane to ensure a continuous margin. In addition, sufficient saline application was performed when using bipolar coagulation forceps. Eighteen months post-surgery, the patient has shown no signs of recurrence. According to the WHO classification, meningiomas are categorized into three grades, with meningothelial, fibrous, and transitional subtypes being the most common in Grade I9). Our patient’s pathology report indicated a meningothelial meningioma, classified as WHO grade 1, with mild cellular atypia.
Since 2018, we have reviewed 12 cases of spinal extradural masses, as summarized in Table 1. Hong et al.9) analyzed 12 cases of spinal epidural meningioma reported from 2002 to 2017. Extradural meningiomas were most commonly located in the thoracic spine, followed by the cervical spine, and predominantly affected female patients. Imaging studies typically showed intraspinal extradural masses with mostly homogeneous enhancement. Most masses exhibited an extradural en plaque lesion, indicative of a dural-based origin, also known as the dural tail sign, resulting in a dumbbell-shaped appearance9). Among the 12 cases listed in Table 1, there were 2 cases of atypical meningioma and 1 case of choroid meningioma; the remaining cases were classified as Grade 1 meningiomas. Thus, the majority of the pathology in extradural meningioma was WHO grade 1, consistent with previous research and a 2018 study published in the Journal of Radiology9).
Previous literature reviews of major spinal meningioma series, including both intra- and extradural subtypes, report surgical recurrence rates of 3% to 7%1). However, the recurrence rate after surgery for extradural meningiomas is four times higher than that for intradural meningiomas5,12). Among the 12 reviewed cases, no recurrence was reported9).

CONCLUSION

Spinal epidural meningiomas are rare, typically presenting as WHO grade 1 tumors. Surgical intervention often results in favorable outcomes, including symptom relief and a low recurrence rate.

CONFLICTS OF INTEREST

No potential conflict of interest relevant to this article was reported.

Fig. 1.
(A) Sagittal magnetic resonance imaging (MRI) showing a T1-enhanced extramedullary mass located at the T1-T5 vertebral levels (longitudinal length: 87.96 mm). (B) Axial MRI showing a T1-enhanced extramedullary mass with spinal cord displacement and compression, extending to the left T2-T3 foramen. (C) Axial MRI depicting an enhanced extramedullary mass with spinal cord displacement and compression, extending into the left T3-T4 foramen.
nerve-2024-00626f1.jpg
Fig. 2.
(A) Sagittal computed tomography (CT) image showing calcification in the right aspect of the mass with no evidence of bony erosion. (B) Axial CT image showing calcification in the right aspect of the mass with no evidence of bony erosion.
nerve-2024-00626f2.jpg
Fig. 3.
(A) Post-thoracic total laminectomy image showing the extradural mass. (B) The surgical field after removal of the meningioma, with the dura mater remaining completely intact.
nerve-2024-00626f3.jpg
Fig. 4.
(A) Histology. Hematoxylin and eosin staining (×200 magnification) depicting the histologic pattern of meningothelial meningiomas, characterized by polygonal cells. (B) Immunohistochemistry shows an ill-defined cytoplasm and positivity for epithelial membrane antigen.
nerve-2024-00626f4.jpg
Fig. 5.
Postoperative spine magnetic resonance imaging (MRI), 1 month later (A) Sagittal MRI showing no enhanced extradural lesion at the T2-T4 level. Fluid collection is observed in the soft tissue from the T1-T5 levels. (B, C) Axial MRI showing no definite enhanced extradural lesion at the T2-T3 and T3-T4 levels.
nerve-2024-00626f5.jpg
Table 1.
Review of previously reported cases of spinal extradural meningioma
References Patient no. Age/Sex Location (level) Clinical presentation CT and MRI findings Pathology Treatment Outcome
N'da et al. (2018)16) 1 55/F T2-T3 Progressive lower-limb weakness Dumbbell-shaped mass, homogeneous contrast enhancement Transitional meningioma T2-T3 laminectomy Full recovery of motor function
Ghanchi et al. (2018)8) 1 40/M T6, L1 Progressive back pain radiating to the left flank and thigh. Two extramedullary masses in the left neural foramina of T6 and L1 WHO grade I meningioma T6, L1 laminectomy Resolution of symptoms
Lai et al. (2018)13) 1 35/M C1-C4 Neck pain, numbness, and stiffness in all four limbs and bilateral upper limb and truncal loss of temperature sensation MRI: extradural soft tissue lesion within the right side of the spinal canal extending from C1 to C4 WHO grade I meningioma Anterior decompression and fusion of the cervical spine with C2 and C3 total laminectomy, C1 and C4 partial laminectomy, and fusion of C2 and C3 Full recovery
Sakamoto et al. (2018)22) 1 57/F  C6-T1 Progressive sensorimotor disturbance in the left upper extremity MRI: enhancing, en-plaque tumor at C6-T1, involving a non-enhancing part, and considerable compression of the spinal cord Fibrous meningioma with metaplastic ossification Hemilaminectomy of C5-C7 Neurological deficits remarkably improved
Tulloch et al. (2020)24) 1 Mild lower limb pyramidal weakness Thoracic spine Extradural chordoid meningioma Successfully resected through a posterior thoracic laminectomy Neurological deficit resolved, no recurrence
Shui et al. (2021)23) 1 66/F T3-T5 Progressive paraparesis Heterogeneously enhancing lesion circumferentially involving the spinal cord from T3 to T5 with left-sided T4/5 foraminal extension WHO grade I meningioma Gross total resection Full recovery except for mild residual left-sided T2-T5 numbness
Garaud et al. (2022)7) 1 44/F C5-C6 Neck pain and paresthesia of the right thumb and index finger Intradural and extradural location of the mass, which enlarged the neuroforamen WHO grade I transitional meningioma  Resection of its intraforaminal and extraforaminal components with reconstruction patch of the dura Symptoms improved moderately
Nguyen et al. (2021)17) 1 22/F C5-C8 Weakness and numbness in both lower limbs MRI: the whole mass was enhanced and showed two portions: intradural and extradural Psammomatous meningioma Completely removed 6 months after surgery, the patient was able to walk with a walker
Pommier et al. (2021)20) 1 74/F Lt.C6-C7 Radicular pain along the left C7 territory MRI: contrast-enhanced tumor inside the left C6-C7 neuroforamen Psammomatous WHO grade I meningioma Unilateral posterior cervical approach; a partial facetectomy, dissection of the tumor Pain did not reappear, and the motor deficit improved
di Bonaventura et al. (2023)4) 1 Young female Sensory disturbance Dorsal epidural sleeve-like mass Atypical meningioma Surgical resection Complete neurological recovery
Gader et al. (2023)6) 1 64/M L3-L4 Progressive cauda equina syndrome MRI: two extradural L3-L4 lesions: one was posterolateral, while the second was anterior. These lesions were hypointense on both T1 and T2-WI, with an important enhancement Grade I metaplasic meningioma Fully removed Motor deficit improved from 3 to 4
Hsieh et al. (2024)10) 1 64/M Lt.C6-C7 Progressive pain in the left shoulder MRI: the mass extended from the left 6/7th cervical to the 7th cervical/1st thoracic neural foramina and reached into the left brachial plexus. T1-weighted images with contrast demonstrated an enhanced mass extending into the left brachial plexus accompanied by compression on the spinal cord Atypical meningioma, grade II meningioma Subtotal resection

CT: computed tomography; MRI: magnetic resonance imaging; WHO: World Health Organization; Lt.: left; WI: weighted imaging; F, female, M: male.

REFERENCES

1. Ben Nsir A, Boughamoura M, Mahmoudi H, Kilani M, Hattab N: Uncommon progression of an extradural spinal meningioma. Case Rep Surg 2014:630876, 2014
crossref pmid pmc
2. Bettaswamy G, Ambesh P, Das KK, Sahu R, Srivastava A, Mehrotra A, et al.: Extradural spinal meningioma: Revisiting a rare entity. J Craniovertebr Junction Spine 7:65-68, 2016
crossref pmid pmc
3. Dagain A, Dulou R, Lahutte M, Dutertre G, Pouit B, Delmas JM, et al.: Extradural spinal meningioma: Case report. Neurochirurgie 55:565-568, 2009
crossref pmid
4. di Bonaventura R, Caccavella VM, Latour K, Rapisarda A, Gessi M, Montano N, et al.: Spinal epidural atypical meningioma: Case report and review of the literature. Acta Neurochir Suppl 135:369-373, 2023
crossref pmid
5. Frank BL, Harrop JS, Hanna A, Ratliff J: Cervical extradural meningioma: Case report and literature review. J Spinal Cord Med 31:302-305, 2008
crossref pmid pmc
6. Gader G, Masmoudi M, Ghedira K, Krifa MI, Chelly I, Zammel I, et al.: Lumbar spine epidural meningioma: Report of a rare case. Spinal Cord Ser Cases 9:53, 2023
crossref pmid pdf
7. Garaud S, Boto J, Egervari K, Vargas MI: Extradural spinal meningioma mimicking a schwannoma: Magnetic resonance imaging findings. Can J Neurol Sci 49:467-469, 2022
crossref pmid
8. Ghanchi H, Hariri OR, Takayanagi A, Li G: Multiple extradural spinal meningiomas in a patient with acquired immunodeficiency syndrome: Case report and literature review. World Neurosurg 117:366-370, 2018
crossref pmid
9. Hong W, Kim ES, Lee Y, Lee K, Koh SH, Song H, et al.: Spinal extradural meningioma: A case report and review of the literature. J Korean Soc Radiol 79:11-17, 2018
crossref pdf
10. Hsieh PC, Lu JC, Huang SC, Toh CH, Kuo HC: Unusual clinical presentation of cervical extradural meningioma detected with neuromuscular ultrasound: A case report. Exp Ther Med 27:205, 2024
crossref pmid pmc
11. Kale A, Akyol C, Keskin E, Aydoğmuş E, Aydın HA, Barut F, et al.: Coexistence of cervico-thoracic extradural en-plaque meningioma with multiple intracranial meningiomas. Neurol Neurochir Pol 48:363-367, 2014
crossref pmid
12. Khayal HB, Abograra A, lashhab M: Extradural spinal meningioma in a nine-year-old girl: A case report and review of the literature. Open Access J Neurol Neurosurg 5:555659, 2017
crossref
13. Lai AL, Salkade PR, Chuah KL, Sitoh YY: Extradural cervical spinal meningioma mimicking malignancy. J Radiol Case Rep 12:1-10, 2018
crossref pmid pmc pdf
14. Messori A, Rychlicki F, Salvolini U: Spinal epidural en-plaque meningioma with an unusual pattern of calcification in a 14-year-old girl: Case report and review of the literature. Neuroradiology 44:256-260, 2002
crossref pmid pdf
15. Milz H, Hamer J: Extradural spinal meningiomas. Report of two cases. Neurochirurgia (Stuttg) 26:126-129, 1983
crossref pmid
16. N'da H, Dauleac C, Toquart A, Afathi M, Meyronet D, Barrey C: Thoracic spine intra- and extradural dumbbell-shaped meningioma: Case report and extensive review of the literature with 21 cases. Neurochirurgie 64:206-210, 2018
crossref pmid
17. Nguyen BQ, Tran DDT, Dang TC, Mai TD, Pham HD, Truong VT: Cervical intra-extradural meningioma with en-plaque, dumbbell-shaped, and an unusual calcified pattern in a young patient. Surg Neurol Int 12:454, 2021
crossref pmid pmc
18. Nong L, Zhou D, Xu N, Du R, Jiang X: Lamina replacement with titanium plate fixation improves spinal stability after total lumbar laminectomy. Comput Methods Biomech Biomed Engin 18:1753-1759, 2015
crossref pmid
19. Plank C, Koller A, Mueller-Mang C, Bammer R, Thurnher MM: Diffusion-weighted MR imaging (DWI) in the evaluation of epidural spinal lesions. Neuroradiology 49:977-985, 2007
crossref pmid pdf
20. Pommier B, Vasiljevic A, Barrey C: Purely intraforaminal meningioma in the cervical spine. World Neurosurg 145:432-433, 2021
crossref pmid
21. Roux FX, Nataf F, Pinaudeau M, Borne G, Devaux B, Meder JF: Intraspinal meningiomas: review of 54 cases with discussion of poor prognosis factors and modern therapeutic management. Surg Neurol 46:458-463; discussion 463-464, 1996
crossref pmid
22. Sakamoto K, Tsutsumi S, Nonaka S, Suzuki T, Ishii H, Ito M, et al.: Ossified extradural en-plaque meningioma of the cervical spine. J Clin Neurosci 50:124-126, 2018
crossref pmid
23. Shui C, Turchini J, Davies M: Purely extradural spinal meningioma: A case report and literature review. Surg Neurol Int 12:431, 2021
crossref pmid pmc
24. Tulloch I, Georges H, Phadke R, Hardwidge C: A thoracic extradural chordoid meningioma: A unique case report and literature review. Br J Neurosurg 34:112-114, 2020
crossref pmid
25. Vargas MI, Abu Eid M, Bogorin A, Beltechi R, Boyer P, Javier RM, et al.: Spinal extradural meningiomas: MRI findings in two cases. J Neuroradiol 31:214-219, 2004
pmid
26. Zevgaridis D, Thomé C: Purely epidural spinal meningioma mimicking metastatic tumor: Case report and review of the literature. Spine (Phila Pa 1976) 27:E403-E405, 2002
crossref pmid
Editorial Office
Department of Neurosurgery, Dankook University Hospital
201, Manghyang-ro, Dongnam-gu, Cheonan-si, Chungcheongnam-do 31116, South Korea
TEL: +0082-41-550-6280   FAX: +82-41-556-0524   
E-mail: spine1225@naver.com , mousehunt7@naver.com
About |  Browse Articles |  Current Issue |  For Authors and Reviewers
Copyright © The Korean Society of Peripheral Nervous System.                 Developed in M2PI