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Open access

Michel Gustavo Mondragón-Soto, Luis Del Valle, José Alfredo González-Soto, and Roberto Alfonso De Leo-Vargas

BACKGROUND

Primary intracranial rhabdomyosarcoma is an extraordinarily rare malignant tumor, with even fewer presenting with distant metastasis. To date, only five cases, including the one presented here, have been reported to present metastatic activity.

OBSERVATIONS

A 12-year-old boy presented with a few days of headache, nausea, vomiting, but no neurological deficit. Brain computed tomography and magnetic resonance imaging demonstrated hydrocephalus and a cystic lesion with left parieto-occipital extension. After resection, pathology reported primary rhabdomyosarcoma, with positive desmin and myogenin on immunohistochemistry. The patient presented with pulmonary metastasis. The patient had an overall survival of 21 months after diagnosis with optimal treatment.

LESSONS

Rhabdomyosarcoma is a malignant neoplasm arising from undifferentiated skeletal muscle cells, with morphological, immunohistochemical, ultrastructural, or molecular genetic evidence of primary skeletal muscle differentiation. It presents with a rapidly worsening clinical course and the final outcome is poor. Treatment is widely based on protocols that have been proven to be effective in extracranial versions of these tumors, although repeatedly ineffective. Primary brain rhabdomyosarcoma poses a diagnostic challenge because of its infrequent presentation, grade of undifferentiation and tumor heterogeneity. Immunohistochemical and genetic testing have proven to be useful tools for diagnosis.

Open access

Oliver Y. Tang, Patricia Zadnik Sullivan, Teddi Tubre, Joshua Feler, Belinda Shao, Jesse Hart, and Ziya L. Gokaslan

BACKGROUND

Tumoral calcinosis is an uncommon disease resulting from dystrophic calcium phosphate crystal deposition, with only 7% of cases involving the spine, and it may diagnostically mimic neoplasms.

OBSERVATIONS

In this case, a 54-year-old woman with history of systemic scleroderma presented with 10 months of progressive left lumbosacral pain. Imaging revealed an expansile, 4 × 7-cm, well-circumscribed mass in the lumbosacral spine with L5–S1 neuroforaminal compression. Because intractable pain and computed tomography (CT)-guided needle biopsy did not entirely rule out malignancy, operative management was pursued. The patient underwent L4–S2 laminectomies, left L5–S1 facetectomy, L5 and S1 pediculectomies, and en bloc resection, performed under stereotactic CT-guided intraoperative navigation. Subsequently, instrumented fusion was performed with L4 and L5 pedicle screws and S2 alar-iliac screws. Pathological examination was consistent with tumoral calcinosis, with multiple nodules of amorphous basophilic granular calcified material lined by histiocytes. There was no evidence of recurrence or neurological deficits at 5-month follow-up.

LESSONS

Because spinal tumoral calcinosis may mimic neoplasms on imaging or gross intraoperative appearance, awareness of this clinical entity is essential for any spine surgeon. A review of all case reports of lumbosacral tumoral calcinosis (n = 14 from 1952 to 2016) was additionally performed. The case featured in this report presents the first known case of navigation-assisted resection of lumbosacral tumoral calcinosis.

Open access

Gaston Camino-Willhuber, Ryan S. Beyer, Matthew J. Hatter, Austin J. Franklin, Nolan J. Brown, Sohaib Hashmi, Michael Oh, Nitin Bhatia, and Yu-po Lee

BACKGROUND

Pyogenic spinal infections (PSIs) are a group of uncommon but serious infectious diseases that are characterized by inflammation of the endplate–disc unit. PSIs are considered more prevalent and aggressive among patients with chronic immunocompromised states. Association between PSIs and liver disease has not been systematically analyzed. The authors performed a systematic review to study baseline characteristics, clinical presentation, and mortality of patients with PSI in the setting of chronic liver disease.

OBSERVATIONS

The authors presented the case of a 72-year-old female patient with chronic liver disease who presented with severe low back pain and bilateral lower weakness. Imaging studies showed T10–11 spondylodiscitis. The patient received decompression and fusion surgery with partial neurological improvement. The authors performed a systematic literature search of spondylodiscitis and liver disease, and eight published articles met the studies inclusion and exclusion criteria. These studies featured a total of 144 patients, of whom 129 met inclusion criteria (mean age, 60.5 years, range 40 to 83 years; 62% males). Lumbar infection was the most common report (67%), with Staphylococcus aureus (48%) as the main causative microorganism. Neurological compromise was present in 69% of patients. Surgical intervention occurred in 70.5% of patients, and the average duration of antibiotic treatment was 69.4 days. Postoperative complication rate was 28.5%, with a 30- and 90-day mortality of 17.2% and 24.8%, respectively.

LESSONS

Pyogenic spondylodiscitis in patients with liver disease was associated with a high rate of neurological compromise, postoperative complications, and mortality.

Open access

Hiroshi Baba, Hiro Kiyosue, Satomi Ide, Kouhei Onishi, Takeshi Kubo, and Kohei Tokuyama

BACKGROUND

Although osseous involvement is occasionally observed in spinal epidural arteriovenous fistulas (AVFs) or seen as a part of diseases of spinal arteriovenous metameric syndrome, purely intraosseous spinal AVFs are extremely rare. Their clinical and imaging characteristic features are not well known. The authors present a case of purely intraosseous AVFs associated with compression fracture.

OBSERVATIONS

A 76-year-old man presented with back pain and progressive myelopathy. Computed tomography showed compression fracture of the T12 vertebral body and dilatation of perimedullary veins. Spinal angiography revealed an intraosseous AVF at the T12 spine level, which was fed by multiple feeders of ventral somatic branches and drained into the paravertebral and perimedullary veins. The intraosseous AVF was completely occluded by the combined techniques of transarterial and transvenous embolization with glue and a coil. The symptoms disappeared within 1 month after embolization.

LESSONS

Although extremely rare, spinal intraosseous AVFs can develop after compression fracture and cause congestive myelopathy. Combined transarterial and transvenous embolization is useful for the specific case of spinal intraosseous AVFs with both paravertebral and perimedullary drainage.

Open access

Ryan S. Beyer, Austin J. Franklin, Matthew J. Hatter, Andrew Nguyen, Nolan J. Brown, Gaston Camino-Willhuber, Nestor R. Davies, Sohaib Hashmi, Michael Oh, Nitin Bhatia, and Yu-Po Lee

BACKGROUND

Primary spinal infections (PSIs) are a group of uncommon but serious infectious diseases that are characterized by inflammation of the endplate-disc unit. Pediatric spinal infection is rare and challenging to diagnose due to vague presenting symptoms. Most cases are conservatively managed with surgery rarely indicated. The authors performed a systematic review to study the baseline characteristics, clinical presentation, and outcomes of pediatric patients with PSIs who underwent surgical treatment.

OBSERVATIONS

PSI in pediatric patients might behave differently in terms of epidemiology, clinical presentation, and outcomes when compared with nonpediatric patients. Overall, PSI ultimately managed surgically in pediatric patients is associated with a high rate of localized pain, neurological compromise, and treatment failure when compared with nonsurgically managed pediatric spinal infections.

LESSONS

PSIs managed surgically in the pediatric population were found to be caused by Mycobacterium tuberculosis in 74.4% of cases and were associated with higher rates of localized pain, neurological compromise, and treatment failure than nonsurgically managed pediatric spinal infections. Thoracic involvement (71.8%) in the spinal infection was reported most commonly in our review. When omitting the cases involving M. tuberculosis infection, it was revealed that 50% of the pediatric cases involved infection in the cervical region, suggesting increased severity and disease course of cervical spinal infections in the pediatric population. Surgical treatment is indicated only in cases of severe neurological compromise and treatment failure.

Restricted access

Keita Shibahashi, Hiroyuki Ohbe, and Hideo Yasunaga

OBJECTIVE

Adjuvant medical treatment to reduce the recurrence rate after burr hole surgery for chronic subdural hematoma (CSDH) has not yet been established. This study aimed to investigate the association between tranexamic acid (TXA) use after burr hole surgery and the reoperation rate in patients with CSDH.

METHODS

This observational study used the Japanese Diagnostic Procedure Combination inpatient database, a nationwide inpatient database in Japan, from July 1, 2010, to March 31, 2019. The authors identified patients who were hospitalized for CSDH and underwent burr hole surgery within 2 days of admission. The primary outcome measure was reoperation within 1 year after surgery. One-to-one propensity score–matched analysis was performed to compare the outcomes between patients who started oral TXA within 2 days after surgery (TXA users) and those who did not (TXA nonusers). Robustness of the analyses was assessed using the instrumental variable analysis.

RESULTS

Of the 149,543 patients with CSDH treated at 1100 hospitals, 7366 (4.9%) were TXA users. Propensity score matching created 6564 matched pairs with highly balanced baseline characteristics. The reoperation rate was significantly lower in TXA users than in nonusers (1.9% vs 6.1%, p < 0.001) with a risk difference of −4.1% (95% CI −4.8% to −3.4%). There was no significant difference in composite adverse events (0.6% vs 0.5%, p = 0.817). Total hospitalization costs were also significantly lower in TXA users than in nonusers ($5229 vs $5344 [USD], p < 0.001). The results of the instrumental variable analysis were consistent with those of the propensity score–matched analysis.

CONCLUSIONS

Findings of this study, using a nationwide inpatient database, suggest that adjuvant TXA use after burr hole surgery was associated with a reduced reoperation rate in patients with CSDH.

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Giuseppe Cinalli, Carmela Russo, Francesca Vitulli, Raffaele Stefano Parlato, Pietro Spennato, Alessia Imperato, Mario Quarantelli, Eugenio Covelli, and Ferdinando Aliberti

OBJECTIVE

The authors’ objective was to measure the effect of posterior cranial vault distraction (PCVD) plus foramen magnum decompression (FMD) on dural sinus volume and venous flow in patients with syndromic craniosynostosis.

METHODS

The volumes of the sagittal, straight, transverse, and sigmoid sinuses of 5 consecutive patients with syndromic craniosynostosis who underwent PCVD+FMD were calculated in cubic centimeters with T2-weighted volumetric MRI sequences before surgery, immediately after surgery, and after the end of the distraction process. Tridimensional reconstructions of phase-contrast magnetic resonance angiography (PC-MRA) images were obtained with multiplanar reconstruction (MPR).

RESULTS

The average total volume of all dural sinuses increased immediately after surgery (from 10.06 cm3 to 12.64 cm3) and continued to increase throughout the 30-day distraction period (from 12.64 cm3 to 14.71 cm3) (p = 0.04), except that the right sigmoid sinus remained stable after the initial increase. The most important increases were observed for the left transverse sinus (+113.2%), right transverse sinus (+104.3%), left sigmoid sinus (+91.3%), and sagittal sinus (+41.8%). Less important modifications were evident for the right sigmoid sinus (+33.7%) and straight sinus (+23.4%). Significant improvements in venous flow were noted on the tridimensional reconstructions of the PC-MRA images. Venous obstruction grading score improved in 4 patients (average [range] 2.4 [ 2–5]) (p = 0.023) and remained stable in 1 patient. All patients had chronic tonsillar herniation (CTH) (mean [range] 16.6 [8–26] mm), and 3 had syringomyelia. CTH showed improvement on the last follow-up MRI evaluation in 4 patients (mean [range] 10.5 [0–25] mm) and worsened from 15 mm to 19 mm in 1 patient. Syringomyelia improved in 2 patients and remained unchanged in 1.

CONCLUSIONS

This study has provided the first radiological evidence of the impact of craniofacial surgery on dural sinus anatomy and venous drainage. The venous anomalies described in patients with syndromic craniosynostosis are not static, and PCVD+FMD triggers a dynamic process that can lead to significant modifications of intracranial venous drainage. The traction exerted by the distracted bone flap onto the occipitoparietal dura mater adherent to the inner calvaria may account for the enlargement of the dural sinus throughout the distraction period. The impact of these modifications on venous pressure, intracranial pressure, CTH, and hydrocephalus remains to be determined.

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Ananth P. Abraham, Tarang K. Vora, Bernice T. Selvi, and Vedantam Rajshekhar

OBJECTIVE

The aims of this study were to characterize syringomyelia in patients with lipomyelomeningocele (LMMC) and investigate the association between the presence of a syrinx and patient neurological deficits.

METHODS

Patients with LMMC who had been admitted between 2007 and 2020 were included in the study. Syringomyelia on magnetic resonance imaging (MRI) was classified into three groups: true syrinx, early syrinx, and presyrinx. The correlation of clinical deficits (at birth, new onset, and progressive) and type of lipoma with the presence and type of syrinx was studied.

RESULTS

Among a total of 140 patients, the median age was 2.5 (IQR 1.3–9) years. Neurological deficits were present at birth in 75 (53.6%) patients, but 67 (47.9%) had new-onset deficits or progression of their birth deficits. The most common type of spinal lipoma was the transitional type (75.7%). Thirty-nine (27.9%) patients had a syrinx (37 with a true syrinx, 2 with an early syrinx), and 25 (17.9%) patients had a presyrinx. There was no significant correlation between the presence of a syrinx and patient neurological deficits. However, patients with a syrinx that was not adjacent to the lipoma (≥ 1 vertebral level away) had a higher incidence of deficits at birth (p = 0.045), whereas those with a syrinx spanning ≥ 5 vertebral levels had a higher incidence of progressive neurological deficits (p = 0.04). Six (75%) of 8 patients in whom serial MRI had shown syrinx progression had clinical worsening.

CONCLUSIONS

Syrinx location with respect to the spinal lipoma, syrinx length, and progressive syringomyelia may have clinical significance in patients with LMMC.

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Yeong Jin Kim, Kyung-Sub Moon, Woo-Youl Jang, Tae-Young Jung, In-Young Kim, and Shin Jung

OBJECTIVE

Tuberculum sellae meningiomas (TSMs) present a burdensome surgical challenge because of their adjacency to vital neurovascular structures. The contralateral subfrontal approach provides an outstanding corridor for removing a TSM with an excellent visual outcome and limited complications. The authors present their long-term surgical experience in treating TSMs via the contralateral subfrontal approach and discuss patient selection, surgical techniques, and clinical outcomes.

METHODS

Between 2005 and 2021, the authors used the contralateral subfrontal approach in 74 consecutive patients presenting with TSMs. The surgical decision-making process and surgical techniques are described, and the clinical outcomes were retrospectively analyzed.

RESULTS

The mean patient age was 54.4 years, with a female predominance (n = 61, 82%). Preoperatively, 61 patients (82%) had vision symptoms and 73 (99%) had optic canal invasion by tumor. Gross-total resection was achieved in almost all patients (n = 70, 95%). The visual function improvement and stabilization rate was 91% (67/74). Eight patients (11%) showed a worsening of visual function on the less-compromised (approach-side) optic nerve. There was no occurrence of cerebrospinal fluid leakage. Four patients (5%) experienced recurrences after the initial operation (mean follow-up duration 63 months). There were no deaths in this study.

CONCLUSIONS

The contralateral subfrontal approach provides a high chance of complete tumor removal and visual improvement with limited complications and recurrences, especially when the tumor is in a unilateral or midline location causing unilateral visual symptoms or bilateral asymmetrical visual symptoms, regardless of tumor size or encasement of major vessels. With the appropriate patient selection, surgical technique, and familiarity with surrounding neurovascular structures, this approach is reliable for TSM surgery.

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Chongwon Pae, Myung Ji Kim, Won Seok Chang, Hyun Ho Jung, Kyung Won Chang, Jinseok Eo, Hae-Jeong Park, and Jin Woo Chang

OBJECTIVE

Thalamotomy at the nucleus ventralis intermedius using MR-guided focused ultrasound has been an effective treatment method for essential tremor (ET). However, this is not true for all cases, even for successful ablation. How the brain differs in patients with ET between those with long-term good and poor outcomes is not clear. To analyze the functional connectivity difference between patients in whom thalamotomy was effective and those in whom thalamotomy was ineffective and its prognostic role in ET treatment, the authors evaluated preoperative resting-state functional MRI in thalamotomy-treated patients.

METHODS

Preoperative resting-state functional MRI data in 85 patients with ET, who were experiencing tremor relief at the time of treatment and were followed up for a minimum of 6 months after the procedure, were collected for the study. The authors conducted a graph independent component analysis of the functional connectivity matrices of tremor-related networks. The patients were divided into thalamotomy-effective and thalamotomy-ineffective groups (thalamotomy-effective group, ≥ 50% motor symptom reduction; thalamotomy-ineffective group, < 50% motor symptom reduction at 6 months after treatment) and the authors compared network components between groups.

RESULTS

Seventy-two (84.7%) of the 85 patients showed ≥ 50% tremor reduction from baseline at 6 months after thalamotomy. The network analysis shows significant suppression of functional network components with connections between the areas of the cerebellum and the basal ganglia and thalamus, but enhancement of those between the premotor cortex and supplementary motor area in the noneffective group compared to the effective group.

CONCLUSIONS

The present study demonstrates that patients in the noneffective group have suppressed functional subnetworks in the cerebellum and subcortex regions and have enhanced functional subnetworks among motor-sensory cortical networks compared to the thalamotomy-effective group. Therefore, the authors suggest that the functional connectivity pattern might be a possible predictive factor for outcomes of MR-guided focused ultrasound thalamotomy.