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Open access

Central nervous system mature teratoma producing carbohydrate antigen 19-9: illustrative case

Shu Takeuchi, Yoshiki Arakawa, Yasuhide Takeuchi, Sachiko Minamiguchi, Masahiro Tanji, Yohei Mineharu, Hironori Haga, and Susumu Miyamoto

BACKGROUND

Central nervous system (CNS) mature teratoma is a rare disease with symptoms that can vary according to tumor location. Most lesions are benign; rarely, malignancy can develop in any of the somatic components. Elevated levels of tumor markers such as α-fetoprotein and β-human chorionic gonadotropin are not usually found in patients with CNS mature teratoma, and no reports have described an association with carbohydrate antigen 19-9 (CA19-9).

OBSERVATIONS

A 64-year-old woman with headache was found to have a mass lesion in the anterior cranial fossa. Magnetic resonance imaging of the brain suggested a mature teratoma. Serum and cerebrospinal fluid (CSF) tests showed significant CA19-9 elevations (2,770 U/mL and 4,387 U/mL, respectively). Other examinations, including whole-body 18F-fluorodeoxyglucose positron emission tomography, did not detect the origin of elevated CA19-9, suggesting that the high CA19-9 levels were caused by intracranial tumor. The patient underwent tumor removal. The histopathological diagnosis was mature teratoma with positive CA19-9 staining. CA19-9 levels in serum and CSF decreased significantly after tumor removal.

LESSONS

The histopathological findings and postoperative decreased CA19-9 levels established the diagnosis of CA19-9–producing CNS mature teratoma. CNS mature teratoma can cause elevations in CA19-9 in cases with absence of neoplasms in the trunk.

In Journal of Neurosurgery: Case Lessons Volume 4 (2022): Issue 7 (Aug 2022)

Open access

Gross-total resection of a suprasellar with recurrent cerebellopontine angle region craniopharyngioma by endoscopic far-lateral supracerebellar infratentorial approach: illustrative case

Tao Xie and Xiaobiao Zhang

BACKGROUND

Craniopharyngiomas that rarely extend into the posterior fossa are treated with staged operations or combined approaches. The authors reported a patient undergoing gross-total resection of a suprasellar with recurrent cerebellopontine angle (CPA) craniopharyngioma using an endoscopic far-lateral supracerebellar infratentorial approach (EFL-SCITA).

OBSERVATIONS

The patient was a 15-year-old boy who presented with headache and decreased vision that lasted for half a year. He previously received three surgeries related to CPA craniopharyngioma. Preoperative magnetic resonance imaging revealed a suprasellar with recurrent CPA craniopharyngioma. Gross-total resection of this suprasellar and CPA tumor was achieved through EFL-SCITA. All symptoms and signs were improved. There were no postsurgical complications except for mild facial paralysis.

LESSONS

EFL-SCITA can be used not only for tumors in the posterolateral pontomesencephalon and ptero-clival-tentorial area but also for tumors in the suprasellar region with posterior fossa extension.

In Journal of Neurosurgery: Case Lessons Volume 4 (2022): Issue 7 (Aug 2022)

Open access

Nature or nurture: a latent ossification of the posterior longitudinal ligament after atlantoaxial fusion. Illustrative case

Shih-Wei Tzeng, Yi-Hsuan Kuo, Chao-Hung Kuo, Hsuan-Kan Chang, Chin-Chu Ko, Tsung-Hsi Tu, Chih-Chang Chang, Henrich Cheng, Wen-Cheng Huang, and Jau-Ching Wu

BACKGROUND

The natural history of ossification of the posterior longitudinal ligament (OPLL) remains poorly understood and multiple etiologies have been reported. However, most have focused on the characteristics of the patient rather than alternation of mechanical stress after spinal fusion.

OBSERVATIONS

This report describes, for the first time, a de novo OPLL found at the subaxial cervical spine 7 years after an atlantoaxial fusion surgery. A 57-year-old female initially required atlantoaxial arthrodesis for os odontoideum and stenosis that caused myelopathy. The posterior fusion surgery went smoothly without complications and the patient had good recovery of neurological functions. There was no associated instability, trauma, or reoperations during the follow-up. Seven years later, the patient presented with slight neck pain and a newly developed OPLL at C3–4 caudal to the C1–2 fusion construct.

LESSONS

Conflicting with the conventional concept that OPLL is common in elderly men with genetic or hormonal factors, or associated spondyloarthropathies, OPLL could develop in women even after solid C1–2 fusion. The adjacent subaxial cervical spine is not free of risks for subsequent development of OPLL and cervical spondylotic myelopathy. This case illustration extends the scope of etiologies of OPLL within the present literature.

In Journal of Neurosurgery: Case Lessons Volume 4 (2022): Issue 7 (Aug 2022)

Open access

Surgical management of high-grade lumbar spondylolisthesis associated with Hajdu-Cheney syndrome: illustrative case

Davaine J. Ndongo Sonfack, David Bergeron, Zhi Wang, Ghassan Boubez, Daniel Shedid, and Sung-Joo Yuh

BACKGROUND

Hajdu-Cheney syndrome (HCS) is a rare connective tissue disorder characterized by severe bone demineralization. In the spine, it is associated with the early onset of severe osteoporosis and can cause spondylolisthesis. Spinal instrumentation in the setting of severe osteoporosis is challenging because of poor resistance of vertebrae to biomechanical stress.

OBSERVATIONS

A 59-year-old woman with known idiopathic HCS presented with a grade 4 L5-S1 spondylolisthesis and right L5 pedicle fracture associated with a left L5 pars fracture, causing a progressive L5 radiculopathy that was worse on the left side than the right side and bilateral foot drop. The authors performed decompressive lumbar surgery, which included a complete L5 laminectomy and resection of the left L5 pedicle. This was followed by multilevel lumbosacral instrumentation using cement-augmented fenestrated pedicle screws as well as transdiscal sacral screws and bilateral alar-iliac fixation. Postoperatively, the radicular pain resolved, and the left foot drop partially recovered.

LESSONS

Stabilization of high-grade spondylolisthesis in the setting of bone demineralization disorders is challenging. The use of different instrumentation techniques is important because it increases biomechanical stability of the overall instrumentation construct.

In Journal of Neurosurgery: Case Lessons Volume 4 (2022): Issue 7 (Aug 2022)

Open access

Total en bloc spondylectomy for the L5 metastasis of a carcinoid tumor: illustrative case

Cristina P. Jódar, Simón Fuentes Caparrós, Miguel A. Marín, and Julio Osuna Soto

BACKGROUND

Total en bloc spondylectomy (TES) was designed to achieve oncological complete tumor resection in a vertebral compartment. Because of the special anatomy of the lumbosacral junction, TES procedure at the L5 level is a challenge, and it has been explained in few reports in the literature. Performing TES in the lower lumbar region, as normal, is accomplished by using a combined approach.

OBSERVATIONS

The authors presented the case of a 20-year-old man with an isolated spinal metastasis at the L5 level of carcinoid tumor of jejunum, limited to the vertebral body. Due to good long-term prognosis, after multidisciplinary evaluation the authors decided to treat the patient with TES through a combined posteroanterior approach, with posterior instrumentation and anterior reconstruction. Nine years after surgery, the patient was asymptomatic, with no sign of local recurrence.

LESSONS

TES is a feasible technique to provide long-term survival in a select subgroup of patients, reducing the risk of local recurrence. The authors presented some anatomical and biomechanical factors that must be considered at the lumbosacral region. Despite the high rates of complication associated with TES, most patients benefit from local control provided by the technique.

In Journal of Neurosurgery: Case Lessons Volume 4 (2022): Issue 7 (Aug 2022)

Restricted access

Does displacement of cervical and thoracolumbar dislocation-translation injuries predict spinal cord injury or recovery?

Mark J. Lambrechts, Nicholas D. D’Antonio, Brian A. Karamian, Arun P. Kanhere, Azra Dees, Bright M. Wiafe, Jose A. Canseco, Barrett I. Woods, I. David Kaye, Jeffrey Rihn, Mark Kurd, Alan S. Hilibrand, Christopher K. Kepler, Alexander R. Vaccaro, and Gregory D. Schroeder

OBJECTIVE

For patients with cervical and thoracolumbar AO Spine type C injuries, the authors sought to 1) identify whether preoperative vertebral column translation is predictive of a complete spinal cord injury (SCI) and 2) identify whether preoperative or postoperative vertebral column translation is predictive of neurological improvement after surgical decompression.

METHODS

All patients who underwent operative treatment for cervical and thoracolumbar AO Spine type C injuries at the authors’ institution between 2006 and 2021 were identified. CT and MRI were utilized to measure vertebral column translation in millimeters prior to and after surgery. A receiver operating characteristic (ROC) curve was generated to predict the probability of sustaining a complete SCI on the basis of the amount of preoperative vertebral column translation. ROC curves were then used to predict the probability of neurological recovery on the basis of preoperative and postoperative vertebral column translation.

RESULTS

ROC analysis of 67 patients identified 6.10 mm (area under the curve [AUC] 0.77, 95% CI 0.650–0.892) of preoperative vertebral column translation as predictive of complete SCI. Additionally, ROC curve analysis found that 10.4 mm (AUC 0.654, 95% CI 0.421–0.887) of preoperative vertebral column translation was strongly predictive of no postoperative neurological improvement. Residual postoperative vertebral column translation after fracture reduction and instrumentation had no predictive value on neurological recovery (AUC 0.408, 95% CI 0.195–0.622).

CONCLUSIONS

For patients with cervical and thoracolumbar AO Spine type C injuries, the amount of preoperative vertebral column translation is highly predictive of complete SCI and the likelihood of postoperative neurological recovery.

In Journal of Neurosurgery: Spine Publish Before Print

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Editorial. Neurosurgical capacity-building in Africa: how do we build an equitable future?

Cletus Cheyuo and Mojgan Hodaie

In Journal of Neurosurgery Publish Before Print

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Equity in neurosurgical scientific publication

William W. Ashley Jr., Sonia V. Eden, and James T. Rutka

In Journal of Neurosurgery Publish Before Print

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Etiology- and region-specific characteristics of transependymal cerebrospinal fluid flow

Peter H. Yang, Alison Almgren-Bell, Hongjie Gu, Anna V. Dowling, Sangami Pugazenthi, Kimberly Mackey, Esther B. Dupépé, and Jennifer M. Strahle

OBJECTIVE

Transependymal flow (TEF) of CSF, often delineated as T2-weighted hyperintensity adjacent to the lateral ventricles on MRI, is a known imaging finding, usually in the setting of CSF flow disturbances. Specific radiological features of TEF and their relationships with clinical markers of hydrocephalus and underlying disease pathology are not known. Here, the authors describe the radiological features and clinical associations of TEF with implications for CSF circulation in the setting of intracranial pathology.

METHODS

After obtaining IRB review and approval, the authors reviewed the radiological records of all patients who underwent intracranial imaging with CT or MRI at St. Louis Children’s Hospital, St. Louis, Missouri, between 2008 and 2019 to identify individuals with TEF. Then, under direct review of imaging, TEF pattern, degree, and location and underlying pathology and other radiological and clinical features pertaining to CSF circulation and CSF disturbances were noted.

RESULTS

TEF of CSF was identified in 219 patients and was most prevalent in the setting of neoplasms (72%). In 69% of the overall cohort, TEF was seen adjacent to the anterior aspect of the frontal horns and the posterior aspect of the occipital horns of the lateral ventricles, and nearly half of these patients also had TEF dorsal to the third ventricle near the splenium of the corpus callosum. This pattern was independently associated with posterior fossa medulloblastoma when compared with pilocytic astrocytoma (OR 4.75, 95% CI 1.43–18.53, p = 0.0157). Patients with congenital or neonatal-onset hydrocephalus accounted for 13% of patients and were more likely to have TEF circumferentially around the ventricles without the fronto-occipital distribution. Patients who ultimately required permanent CSF diversion surgery were more likely to have the circumferential TEF pattern, a smaller degree of TEF, and a lack of papilledema at the time of CSF diversion surgery.

CONCLUSIONS

CSF transmigration across the ependyma is usually restricted to specific periventricular regions and is etiology specific. Certain radiological TEF characteristics are associated with tumor pathology and may reflect impaired or preserved ependymal fluid handling and global CSF circulation. These findings have implications for TEF as a disease-specific marker and in understanding CSF handling within the brain.

In Journal of Neurosurgery: Pediatrics Publish Before Print

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Evaluating risk of recurrence in patients with meningioma

Jeffrey I. Traylor, Aaron R. Plitt, William H. Hicks, Tabarak M. Mian, Bruce E. Mickey, and Samuel L. Barnett

OBJECTIVE

Meningioma prognostication and treatment continues to evolve with an increasing understanding of tumor biology. In this study, the authors aimed to test conventional predictors of meningioma recurrence, histopathology variables for which there exists some controversy (brain invasion), as well as a novel molecular-based location paradigm.

METHODS

This is a retrospective study of a consecutive series of patients with WHO grade I–III meningioma resected at The University of Texas Southwestern Medical Center between 1994 and 2015. Time to meningioma recurrence (i.e., recurrence-free survival [RFS]) was the primary endpoint measured. Kaplan-Meier curves were constructed and compared using log-rank tests. Cox univariate and multivariate analyses were performed to identify predictors of RFS.

RESULTS

A total of 703 consecutive patients with meningioma underwent resection at The University of Texas Southwestern Medical Center between the years 1994 and 2015. A total of 158 patients were excluded for insufficient follow-up (< 3 months). The median age of the cohort was 55 years (range 16–88 years) and 69.5% (n = 379) were female. The median follow-up was 48 months (range 3–289 months). There was not a significantly increased risk of recurrence in patients with evidence of brain invasion, in patients with otherwise WHO grade I meningioma (Cox univariate HR 0.92, 95% CI 0.44–1.91, p = 0.82, power 4.4%). Adjuvant radiosurgery to subtotally resected WHO grade I meningiomas did not prolong the time to recurrence (n = 52, Cox univariate HR 0.21, 95% CI 0.03–1.61, p = 0.13, power 71.6%). Location (midline skull base, lateral skull base, and paravenous) was significantly associated with RFS (p < 0.01, log-rank test). In patients with high-grade meningiomas (WHO grade II or III), location was predictive of RFS (p = 0.03, log-rank test), with paravenous meningiomas exhibiting the highest rates of recurrence. Location was not significant on multivariate analysis.

CONCLUSIONS

The data suggest that brain invasion does not increase the risk of recurrence in otherwise WHO grade I meningioma. Adjuvant radiosurgery to subtotally resected WHO grade I meningiomas did not prolong the time to recurrence. Location categorized by distinct molecular signatures did not predict RFS in a multivariate model. Larger studies are needed to confirm these findings.

In Journal of Neurosurgery Publish Before Print