Upper cervical intramedullary schwannoma of the spinal cord presenting with myelopathy: illustrative case

Shyam Duvuru Department of Neurosurgery, Apollo Specialty Hospitals, Madurai, Tamil Nadu, India

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Vivek Sanker Department of Neurosurgery, Trivandrum Medical College, Kerala, India

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Naureen Syed Department of Clinical Research, UT MD Anderson Cancer Center, Houston, Texas

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Shubham Mishra Deanery of Clinical Sciences, College of Medicine and Veterinary Medicine, The University of Edinburgh, Edinburgh, United Kingdom

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Sayantika Ghosh Department of Microbiology and Immunology, Georgetown University Medical Center, Washington, DC; and

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Tirth Dave Department of Internal Medicine, Bukovinian State Medical University, Chernivtsi, Ukraine

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BACKGROUND

Intramedullary schwannomas account for 1.1% of all spinal schwannomas. Preoperative diagnosis is best accomplished by thoroughly evaluating clinical and radiological characteristics, accompanied by a high index of suspicion. The authors report a case of C2–3 intramedullary schwannoma in a young male who presented with neck pain and vertigo. The current literature is also reviewed.

OBSERVATIONS

The authors reviewed the data of a young male with a 2-month history of neck pain and vertigo. Magnetic resonance imaging of the brain and cervical spine showed an intramedullary mass at C2–3 with a syrinx extending into the cervicomedullary junction. Laminectomy, myelotomy, and microsurgical excision of the mass under intraoperative neurological monitoring (IONM) were done. Postoperative pathology reported the specimen as a schwannoma.

LESSONS

Gross-total resection of a schwannoma using IONM is the treatment of choice because of the lesion’s benign nature, a better prognosis, and defined cleavage plane. Schwannomas should be included in the differential diagnosis of intramedullary spinal tumors. Because of its progressive nature, early surgery is recommended in symptomatic patients.

ABBREVIATIONS

CNS = central nervous system; CSF = cerebrospinal fluid; CT = computed tomography; CUSA = cavitron ultrasonic surgical aspirator; GTR = gross-total resection; IONM = intraoperative neurological monitoring; MEP = motor evoked potential; MRI = magnetic resonance imaging; SOL = space-occupying lesion; SSEP = somatosensory evoked potentials

BACKGROUND

Intramedullary schwannomas account for 1.1% of all spinal schwannomas. Preoperative diagnosis is best accomplished by thoroughly evaluating clinical and radiological characteristics, accompanied by a high index of suspicion. The authors report a case of C2–3 intramedullary schwannoma in a young male who presented with neck pain and vertigo. The current literature is also reviewed.

OBSERVATIONS

The authors reviewed the data of a young male with a 2-month history of neck pain and vertigo. Magnetic resonance imaging of the brain and cervical spine showed an intramedullary mass at C2–3 with a syrinx extending into the cervicomedullary junction. Laminectomy, myelotomy, and microsurgical excision of the mass under intraoperative neurological monitoring (IONM) were done. Postoperative pathology reported the specimen as a schwannoma.

LESSONS

Gross-total resection of a schwannoma using IONM is the treatment of choice because of the lesion’s benign nature, a better prognosis, and defined cleavage plane. Schwannomas should be included in the differential diagnosis of intramedullary spinal tumors. Because of its progressive nature, early surgery is recommended in symptomatic patients.

ABBREVIATIONS

CNS = central nervous system; CSF = cerebrospinal fluid; CT = computed tomography; CUSA = cavitron ultrasonic surgical aspirator; GTR = gross-total resection; IONM = intraoperative neurological monitoring; MEP = motor evoked potential; MRI = magnetic resonance imaging; SOL = space-occupying lesion; SSEP = somatosensory evoked potentials

Primary tumors of the spinal cord are infrequent and comprise approximately 2% to 10% of all central nervous system (CNS) tumors.1 Typically, these tumors are of a benign nature and are categorized into extradural, intradural-extramedullary, and intramedullary spinal tumors based on their locations. Intramedullary spinal cord tumors are the least common among them all, accounting for 2% to 5% of all spinal tumors. They originate from the glial cells and grow inside the spinal cord, invading and destroying the gray and white matter.2 It has been postulated that the underlying cause of these tumors may be attributed to the presence of small perivascular bundles of peripheral nerves within the spinal cord.3 Few of them have been documented at the level of the conus medullaris, and the majority are in the cervical region.4 As previously reported, 30% of primary intraspinal neoplasms are schwannomas.5 Intramedullary schwannomas account for 0.3% of intraspinal tumors and 1.1% of intraspinal schwannomas.6 However, intramedullary schwannomas are rarely seen and account for 1.1% of all spinal schwannomas.7 The Schwann cell is thought to be the schwannoma’s cell of origin, although it is usually not found in the brain and spinal cord.8

Because of its scarcity among intraspinal tumors and variable clinical presentation, preoperative diagnosis is best accomplished by thoroughly evaluating clinical and radiological characteristics with a high index of suspicion. Spinal magnetic resonance imaging (MRI) is the gold standard for diagnosing intradural spinal tumors. Still, there are instances in which other diagnostic methods, such as computed tomography (CT) scans, cerebrospinal fluid (CSF) analyses, and neurological examinations, can provide valuable insights.9 In cases in which patients experience symptoms, early surgical intervention is recommended to prevent further deterioration of neurological deficits.10 In order to minimize the risk of postoperative complications, the selection of a surgical approach for the management of these tumors should be guided by intraoperative observations such as tumor adhesions.11 Intramedullary schwannomas are exceedingly rare, but the differential diagnoses of intramedullary tumors have very good outcomes when safe gross-total resection (GTR) is completed. Because this is a rare tumor, only case reports are available, and we discuss all the cases that have been published. Diagnosis is established with histopathology, and the origin is still enigmatic. Safe GTR with intraoperative neuromonitoring (IONM) guidance is the treatment of choice, and the role of radiotherapy is dubious. The present report describes the surgical removal of an intramedullary schwannoma from a 23-year-old male together with subsequent histological confirmation.

Illustrative Case

Clinical History

A 23-year-old male presented with a 2-month history of neck pain and vertigo. There was no other preexisting illness. Physical and neurological examinations were within normal limits with no neurological deficits. Routine blood investigations at admission were also within normal limits.

Imaging Examination

MRI of the cervical spine and brain was performed with contrast. Imaging revealed an intramedullary tumor in the cord at the C2–3 level with a syrinx from the lower medulla until C5. The patient was diagnosed with C2–3 intramedullary space-occupying lesion (SOL; Fig. 1).

FIG. 1
FIG. 1

Preoperative sagittal MRI (A–C) showing an intramedullary tumor at the C2–3 level.

Management

After a pre-anesthetic check, the patient underwent C2–4 laminectomy and midline myelotomy while under endotracheal tube intubation general anesthesia. Gross-total excision of the lesion under IONM guidance was performed. The patient was positioned prone with the head held in a Mayfield 3-pin head holder; a midline vertical skin incision was made from C1 to C5. After subperiosteal dissection and muscle retraction, C2–4 laminectomy was performed. Under a microscope, durotomy was performed and retracted followed by a midline myelotomy. The tumor was identified and removed using cavitron ultrasonic surgical aspirator (CUSA), and hemostasis was achieved. The dura mater was closed watertight, and a drain was placed. Throughout the procedure, there was no significant drop in motor evoked potentials (MEPs) and somatosensory evoked potentials (SSEPs). The wound was closed in layers, and a sterile dressing was applied (Fig. 2).

FIG. 2
FIG. 2

Postoperative sagittal MRI (A and B) after GTR.

Utilizing IONM is essential during the resection of such lesions, as it ensures safe and complete removal of the tumor. In this case, we used SSEPs and MEPs for monitoring the patient’s neural function. Additionally, D wave monitoring could be considered in similar scenarios. The implementation of these neuromonitoring techniques allows real-time assessment of neurological integrity during the surgical procedure, minimizing the risk of postoperative neurological deficits and contributing to favorable patient outcomes.

Histopathology and Immunohistochemistry

The tumor appeared soft, well-defined, moderately vascular, and grayish on gross examination. Microscopically, the section showed lesions composed of a biphasic component, Antoni-A, a hypercellular area, and Antoni-B, a hypocellular area. Cellular areas of the specimen exhibited nuclear palisading around the fibrillary process (Verocay bodies). The cells present in these areas were narrow and elongated and displayed a wavy pattern with tapered ends. These cells were interspersed with collagen fibers. Large vessels were seen in the Antoni-B area (Fig. 3). All findings were suggestive of intramedullary cervical schwannoma. Figure 4 presents an intraoperative image demonstrating a well-defined lesion at the C2–3 level, which was debulked using CUSA.

FIG. 3
FIG. 3

Histopathological findings of the schwannoma. Hematoxylin and eosin, original magnification ×400 (left) and ×100 (right).

FIG. 4
FIG. 4

Intraoperative image demonstrating a well-defined lesion at the C2–3 level, which was debulked using a CUSA.

At the 2-year follow up, the patient had symptoms of decreased fine touch and vibration below his neck. There was no weakness or neck pain. His imaging showed a resolving syrinx and no residual or recurrent tumor.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

In this case report, we describe the case of a 23-year-old male patient who was diagnosed with an intramedullary SOL at the C2–3 level. Subsequently, it was revealed that the lesion was an intramedullary schwannoma. Along with neurofibromas, these tumors account for most (30%) primary spinal tumors.12 Based on a study, the rarity of intramedullary schwannoma is evident, as only 70 cases have been reported up to 2018. One possible explanation for this rarity is the lack of Schwann cells within the CNS.5 Typically, intramedullary schwannomas manifest as solitary tumors. However, some cases have also been sporadically associated with neurofibromatosis.7 The male/female ratio of intramedullary schwannoma is approximately 3:1. The most common location of this lesion is the cervical spinal cord (63%), followed by the thoracic (26%) and lumbar (11%) spinal cord.8

Common clinical manifestations of intramedullary schwannoma include sensitivity complaints, sphincter dysfunction, muscle fasciculations, and radicular pain initially, which later progresses to pyramidal and sensory tract dysfunctions.8,13 It is worth noting that many of the clinical features of our patient were classic, which are well documented in previous studies (Table 1). We know that the brain and spinal cord are devoid of Schwann cells; this makes the pathogenesis of intramedullary schwannoma controversial. However, there have been some proposed hypotheses that try to explain its origin.14

TABLE 1

Summary of some of the previous studies on intramedullary schwannoma

Authors & YearAge (yrs)/SexLocationSymptomsProcedure/TreatmentOutcome
Dandpat et al., 20211317/MMedulla–C5Progressive spastic quadriparesis & paraesthesia for 6 mosC1–3 laminoplastyDied after postoperative day 10 due to ventilator-associated pneumonia
Shahab et al., 20221552/MT11Instability in gait & numbness in bilat lower limbsT10 partial, T11 complete, & T12 partial laminectomyNo new motor or sensory deficits in lower limb w/ overall improvement
Decharin et al., 20221663/MC4–6Severe spastic tone, progressive weakness, & numbness in both legs & hands since 5 yrsLaminectomy6-mo postoperative, patient improved, including better handgrip power & ambulation
Tahta et al., 20221738/MCervico-medullary junctionConstant & radiating rt arm & shoulder pain for 1 moCraniotomy & myelotomyPostoperative MRI of brain & cervical spine revealed no residual lesions & symptoms improved gradually
Varshney et al., 20201870/MT11–L2Progressive paraparesis & sphincter dysfunction for 2.5 yrsT12–L1 laminectomyNeurological functions remained unchanged postoperative; 3 mos later, motor function was 4/5 in lt & 3/5 in rt lower extremity & bladder control regained
Dai et al., 20191934/MC3–4Nonradiating neck pain w/ progressive weakness of lt limbs & sensory loss of both lower extremitiesC3–4 laminectomyPostoperative MRI verified successful tumor removal & resolution of cranial spinal cord edema
Navarro Fernández et al., 20182019/MC6–7Pain, numbness, & dysesthesias in rt arm & hand w/ progressive spastic quadriparesisC6–7 laminoplastyAt 4-wk FU, strength recovery (score 4– on MRC scale), increased sensitivity, & modified McCormick score III; postoperative MRI indicated complete tumor removal
Jagannatha et al., 20162111/MT8–9Weakness in both lower limbsT10–12 laminotomyPostoperative progress smooth & significant improvement at 6-mo FU
Li et al., 20172230/MC3–5Lt chest pain & arm weaknessDied before procedure
Gupta et al., 20152348/MC3–4Paraesthesia of all limbs, progressive spastic quadriparesis, & urinary urgency for 5 mosC3–4 laminectomyPostoperative, reduced limb spasticity & improved bilat hand grip strength; at 1-yr FU, notable decrease in paraesthesia & spasticity
Karatay et al., 20171130/FT12–L1Gait disturbance, back pain, & progressive numbness in both legs for 2 mosT12 laminectomyNoticeable improvement observed in neurological exam
Eljebbouri et al., 20132410/MT7–9Bladder & bowel incontinence w/ lower limb weaknessT7–9 laminectomyAt 15-mo FU, neurological recovery; sensation & strength in all limbs returned to normal, but deep tendon reflexes in rt lower limb remained exaggerated
Ohtonari et al., 2009429/MConus medullarisSexual impotence, bladder dysfunction, & paraesthesia in buttocks for 8 mosT12–L1 laminectomyInitially, deterioration in bladder function, but gradually recovered over time
Hayashi et al., 20092578/FT11–L1Pain & numbness in both lower limbsT10–L2 laminectomyPostoperative, transient worsening of lower-extremity weakness & sensory disturbance, but pain improved; JOA score remained at 2 points
Nicácio et al., 20092640/MC4–6Sphincter disturbances & spastic quadriparesisC3–5 laminotomyAt the 24-mo mark, a notable regression of motor & sphincter dysfunction observed

FU = follow-up.

These hypotheses include the presence of ectopic Schwann cells during embryological development,27 the proliferation of Schwann cells ensheathing aberrant intramedullary nerve fibers,28 the extension of Schwann cells along the intramedullary perivascular nervous plexus,29 Schwann cells resulting from the differentiation of primitive multipotential mesenchymal cells,30 and Schwann cells originating from the posterior nerve root at the root entry zone.31

Observations

In our case, the patient presented with a history of neck pain and vertigo. MRI revealed a solitary lesion with a syrinx in the lower medulla to C5 level. An MRI scan is considered the most helpful for diagnosing intramedullary schwannomas. Generally, the tumor demonstrates hypointense signals compared to the spinal cord on T1-weighted scans and hyperintense alerts along with isointense or low signals on T2-weighted scans.7 Differentiating intramedullary schwannoma from astrocytoma or ependymoma can be challenging; however, the presence of an exophytic component related to the nerve root is suggestive of the former. The presence of a thickened enhancing nerve root connected to the tumor is the primary diagnostic indicator on MRI, although it is seldom observed.32 A centrally enhancing lesion accompanied by polar cysts suggests a likelihood of ependymoma.13 Although radiological investigations are often helpful in the preoperative diagnosis, one should always relate clinical and histopathology to prevent the lesion from being misdiagnosed as glioma.5

Lessons

Intramedullary schwannomas are exceedingly rare. In the differential diagnosis of intramedullary tumors, schwannomas are rare but have very good outcomes when safe GTR is completed. Because this is a rare tumor, only case reports are available, and we have discussed all cases that have been published. The diagnosis is established with histopathology, and the origin is still enigmatic. It is mostly seen in young males. MRI is the imaging modality of choice, and the cervical spine is the most common location. Safe GTR with IONM guidance is the treatment of choice, and the role of radiotherapy is dubious.

Author Contributions

Conception and design: Duvuru, Sanker, Syed, Ghosh. Acquisition of data: Duvuru. Analysis and interpretation of data: Duvuru, Sanker, Mishra. Drafting the article: Dave, Sanker, Syed, Mishra, Ghosh. Critically revising the article: Dave, Duvuru, Sanker, Mishra, Ghosh. Reviewed submitted version of manuscript: Dave, Duvuru, Sanker, Ghosh. Approved the final version of the manuscript on behalf of all authors: Dave. Statistical analysis: Duvuru. Administrative/technical/material support: Duvuru. Study supervision: Duvuru, Sanker.

Supplemental Information

Previous Presentations

The abstract was selected for an e-poster submission at the EANS-2023 Conference held in Barcelona, Spain, September 24–28, 2023.

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  • Collapse
  • Expand
  • FIG. 1

    Preoperative sagittal MRI (A–C) showing an intramedullary tumor at the C2–3 level.

  • FIG. 2

    Postoperative sagittal MRI (A and B) after GTR.

  • FIG. 3

    Histopathological findings of the schwannoma. Hematoxylin and eosin, original magnification ×400 (left) and ×100 (right).

  • FIG. 4

    Intraoperative image demonstrating a well-defined lesion at the C2–3 level, which was debulked using a CUSA.

  • 1

    Caro-Osorio E, Herrera-Castro JC, Barbosa-Quintana A, Benvenutti-Regato M. Primary spinal cord small-cell glioblastoma: case report and literature review. World Neurosurg. 2018;118:6970.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2

    M Das J. Hoang S, Mesfin FB. Intramedullary Spinal Cord Tumors. In: StatPearls. StatPearls Publishing; 2023. Accessed July 2, 2023. http://www.ncbi.nlm.nih.gov/books/NBK442031/

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Mukerji G, Sherekar S, Yadav YR, Chandrakar SK, Raina VK. Pediatric intramedullary schwannoma without neurofibromatosis. Neurol India. 2007;55(1):5456.

  • 4

    Ohtonari T, Nishihara N, Ota T, Ota S, Koyama T. Intramedullary schwannoma of the conus medullaris complicated by dense adhesion to neural tissue. Neurol Med Chir (Tokyo). 2009;49(11):536538.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Wood WG, Rothman LM, Nussbaum BE. Intramedullary neurilemoma of the cervical spinal cord. Case report. J Neurosurg. 1975;42(4):465468.

  • 6

    Singh R, Chaturvedi S, Pant I, Singh G, Kumari R. Intramedullary schwannoma of conus medullaris: rare site for a common tumor with review of literature. Spinal Cord Ser Cases. 2018;4(1):99.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Ross DA, Edwards MSB, Wilson CB. Intramedullary neurilemomas of the spinal cord: report of two cases and review of the literature. Neurosurgery. 1986;19(3):458464.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Lodding P, Kindblom LG, Angervall L, Stenman G. Cellular schwannoma: a clinicopathologic study of 29 cases. Vichows Archiv A Pathol Anat. 1990;416(3):237248.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Ottenhausen M, Ntoulias G, Bodhinayake I, et al. Intradural spinal tumors in adults-update on management and outcome. Neurosurg Rev. 2019;42(2):371388.

  • 10

    Yang T, Wu L, Deng X, Yang C, Xu Y. Clinical features and surgical outcomes of intramedullary schwannomas. Acta Neurochir (Wien). 2014;156(9):17891797.

  • 11

    Karatay M, Koktekir E, Erdem Y, Celik H, Sertbas I, Bayar MA. Intramedullary schwannoma of conus medullaris with syringomyelia. Asian J Surg. 2017;40(3):240242.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Ho T, Tai KS, Fan YW, Leong LL. Intramedullary spinal schwannoma: case report and review of preoperative magnetic resonance imaging features. Asian J Surg. 2006;29(4):306308.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Dandpat SK, Tripathi M, Kaur G, Radotra BD, Joshi A, Mohindra S. Cervico medullary junction “intramedullary schwannoma” masquerading as glioma: a surprise during surgery. Neurol India. 2021;69(6):17471752.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Kodama Y, Terae S, Hida K, Chu BC, Kaneko K, Miyasaka K. Intramedullary schwannoma of the spinal cord: report of two cases. Neuroradiology. 2001;43(7):567571.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15

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