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Courtney M. Schusse, Kris Smith and Cornelia Drees

OBJECTIVE

Hemispherectomy is a surgical technique that is established as a standard treatment in appropriately selected patients with drug-resistant epilepsy. It has proven to be successful in pediatric patients with unilateral hemispheric lesions but is underutilized in adults. This study retrospectively evaluated the clinical outcomes after hemispherectomy in adult patients with refractory epilepsy.

METHODS

This study examined 6 cases of hemispherectomy in adult patients at Barrow Neurological Institute. In addition, all case series of hemispherectomy in adult patients were identified through a literature review using MEDLINE and PubMed. Case series of patients older than 18 years were included; reports of patients without clear follow-up duration or method of validated seizure outcome quantification were excluded. Seizure outcome was based on the Engel classification.

RESULTS

A total of 90 cases of adult hemispherectomy were identified, including 6 newly added by Barrow Neurological Institute. Sixty-five patients underwent functional hemispherectomy; 25 patients had anatomical hemispherectomy. Length of follow-up ranged from 9 to 456 months. Seizure freedom was achieved in 80% of patients. The overall morbidity rate was low, with 9 patients (10%) having new or additional postoperative speech or language dysfunction, and 19 patients (21%) reporting some worsening of hemiparesis. No patients lost ambulatory or significant functional ability, and 2 patients had objective ambulatory improvement. Among the 41 patients who underwent additional formal neuropsychological testing postoperatively, overall stability or improvement was seen.

CONCLUSIONS

Hemispherectomy is a valuable surgical tool for properly selected adult patients with pre-existing hemiparesis and intractable epilepsy. In published cases, as well as in this series, the procedure has overall been well tolerated without significant morbidity, and the majority of patients have been rendered free of seizures.

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Alex P. Michael, Matthew W. Weber, Kristin R. Delfino and Venkatanarayanan Ganapathy

OBJECTIVE

While long-term studies have evaluated adjacent-segment disease (ASD) following posterior lumbar spine arthrodesis, no such studies have assessed the incidence and prevalence of ASD following axial lumbar interbody fusion (AxiaLIF). The aim of this study was to estimate the incidence of ASD following AxiaLIF.

METHODS

The authors retrospectively reviewed the medical records of 149 patients who underwent two-level index AxiaLIF and had at least 2 years of radiographic and clinical follow-up. ASD and pre- and postoperative lumbar lordosis were evaluated in each patient. ASD was defined as both radiographic and clinically significant disease at a level adjacent to a previous fusion requiring surgical intervention. The mean duration of follow-up was 6.01 years.

RESULTS

Twenty (13.4%) of the 149 patients developed ASD during the data collection period. Kaplan-Meier analysis predicted a disease-free ASD survival rate of 95.3% (95% CI 90.4%–97.7%) at 2 years and 89.1% (95% CI 82.8%–93.2%) at 5 years for two-level fusion. A laminectomy adjacent to a fusion site was associated with 5.1 times the relative risk of developing ASD. Furthermore, the ASD group had significantly greater loss of lordosis than the no-ASD group (p = 0.033).

CONCLUSIONS

Following two-level AxiaLIF, the rate of symptomatic ASD warranting either decompression or arthrodesis was found to be 4.7% at 2 years and 10.9% at 5 years. Adjacent-segment decompression and postoperative loss of lumbar lordosis predicted future development of ASD. To the authors’ knowledge, this is the largest reported cohort of patients to undergo two-level AxiaLIF in the United States.

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Peyton L. Nisson, Ibrahim Hussain, Roger Härtl, Samuel Kim and Ali A. Baaj

OBJECTIVE

An arachnoid web of the spine (AWS) is a rare and oftentimes challenging lesion to diagnose, given its subtle radiographic findings. However, when left untreated, this lesion can have devastating effects on a patient’s neurological function. To date, only limited case reports and series have been published on this topic. In this study, the authors sought to better describe this lesion, performing a systematic literature review and including 2 cases from their institution’s experience.

METHODS

A systematic literature search was performed in September 2018 that queried Ovid MEDLINE (1946–2018), PubMed (1946–2018), Wiley Cochrane Library: Central Register of Controlled Trials (1898–2018), and Thompson Reuters Web of Science: Citation Index (1900–2018), per PRISMA guidelines. Inclusion criteria specified all studies and case reports of patients with an AWS in which any relevant surgery types were considered and applied. Studies on arachnoid cysts and nonhuman populations, and those that did not report patient treatments or outcomes were excluded from the focus review.

RESULTS

A total of 19 records and 2 patients treated by the senior authors were included in the systematic review, providing a total of 43 patients with AWS. The mean age was 52 years (range 28–77 years), and the majority of patients were male (72%, 31/43). A syrinx was present in 67% (29/43) of the cases. All AWSs were located in the thoracic spine, and all but 2 (95%) were located dorsally (1 ventrally and 1 circumferentially). Weakness was the most frequently reported symptom (67%, 29/43), followed by numbness and/or sensory loss (65%, 28/43). Symptoms predominated in the lower extremities (81%, 35/43). It was found that nearly half (47%, 20/43) of patients had been experiencing symptoms for 1 year or longer before surgical intervention was performed, and 35% (15/43) of reports stated that symptoms were progressive in nature. The most commonly used surgical technique was a laminectomy with intradural excision of the arachnoid web (86%, 36/42). Following surgery, 91% (39/43) of patients had reported improvement in their neurological symptoms. The mean follow-up was 9.2 months (range 0–51 months).

CONCLUSIONS

AWS of the spine can be a debilitating disease of the spine with no more than an indentation of the spinal cord found on advanced imaging studies. The authors found this lesion to be reported in twice as many males than females, to be associated with a syrinx more than two-thirds of the time, and to only have been reported in the thoracic spine; over 90% of patients experienced improvement in their neurological function following surgery.

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Mohammad Sadegh Masoudi, Mohammad Ali Hoghoughi, Fariborz Ghaffarpasand, Shekoofeh Yaghmaei, Maryam Azadegan and Ghazal Ilami

OBJECTIVE

Surgical repair and closure of myelomeningocele (MMC) defects are important and vital, as the mortality rate is as high as 65%–70% in untreated patients. Closure of large MMC defects is challenging for pediatric neurosurgeons and plastic surgeons. The aim of the current study is to report the operative characteristics and outcome of a series of Iranian patients with large MMC defects utilizing the V-Y flap and with latissimus dorsi or gluteal muscle advancement.

METHODS

This comparative study was conducted during a 4-year period from September 2013 to October 2017 in the pediatric neurosurgery department of Shiraz Namazi Hospital, Southern Iran. The authors included 24 patients with large MMC defects who underwent surgery utilizing the bilateral V-Y flap and latissimus dorsi and gluteal muscle advancement. They also retrospectively included 19 patients with similar age, sex, and defect size who underwent surgery using the primary or delayed closure techniques at their center. At least 2 years of follow-up was conducted. The frequency of leakage, necrosis, dehiscence, systemic infection (sepsis, pneumonia), need for ventriculoperitoneal shunt insertion, and mortality was compared between the 2 groups.

RESULTS

The bilateral V-Y flap with muscle advancement was associated with a significantly longer operative duration (p < 0.001) than the primary closure group. Those undergoing bilateral V-Y flaps with muscle advancement had significantly lower rates of surgical site infection (p = 0.038), wound dehiscence (p = 0.013), and postoperative CSF leakage (p = 0.030) than those undergoing primary repair. The bilateral V-Y flap with muscle advancement was also associated with a lower mortality rate (p = 0.038; OR 5.09 [95% CI 1.12–23.1]) than primary closure. In patients undergoing bilateral V-Y flap and muscle advancement, a longer operative duration was significantly associated with mortality (p = 0.008). In addition, surgical site infection (p = 0.032), wound dehiscence (p = 0.011), and postoperative leakage (p = 0.011) were predictors of mortality. Neonatal sepsis (p = 0.002) and postoperative NEC (p = 0.011) were among other predictors of mortality in this group.

CONCLUSIONS

The bilateral V-Y flap with latissimus dorsi or gluteal advancement is a safe and effective surgical approach for covering large MMC defects and is associated with lower rates of surgical site infection, dehiscence, CSF leakage, and mortality. Further studies are required to elucidate the long-term outcomes.

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Adel Elnashar, Smruti K. Patel, Almaz Kurbanov, Kseniya Zvereva, Jeffrey T. Keller and Andrew W. Grande

OBJECTIVE

Percutaneous stereotactic radiofrequency rhizotomy (PSR) is often used to treat trigeminal neuralgia, a serious condition that results in lancinating, episodic facial pain. Thorough understanding of the microsurgical anatomy of the foramen ovale (FO) and its surrounding structures is required for efficient, effective, and safe use of this technique. This morphometric study compares anatomical and surgical orientations to identify the variations of the FO and assess cannulation difficulty.

METHODS

Bilateral foramina from 174 adult human dry skulls (348 foramina) were analyzed using anatomical and surgical orientations in photographs from standardized projections. Measurements were obtained for shape, size, adjacent structures, and morphometric variability effect on cannulation. The risk of potential injury to surrounding structures was also assessed.

RESULTS

The authors identified 6 distinctive shapes of the FO and 5 anomalous variants from the anatomical view, and 6 shapes from the surgical view. In measurements of surface area of this foramen obtained using the surgical view, loss (average 18.5% ± 5.7%) was significant compared with the anatomical view. Morphometrically, foramen size varied significantly and obstruction from a calcified pterygoalar ligament occurred in 7.8% of specimens. Importantly, 8% of foramina were difficult to cannulate, thus posing a 12% risk of inadvertent cannulation of the foramen lacerum.

CONCLUSIONS

Significant variability in the FO’s shape and size probably affected its safe and effective cannulation. Preoperative imaging by 3D head CT may be helpful in predicting ease of cannulation and in guiding treatment decisions, such as a percutaneous approach over microvascular decompression or radiosurgery.

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Fengming Lan, Qing Qin, Huiming Yu and Xiao Yue

OBJECTIVE

Although glucose metabolism reengineering is a typical feature of various tumors, including glioma, key regulators of glycolytic reprogramming are still poorly understood. The authors sought to investigate whether glycolysis inhibition by microRNA (miR)–448 increases radiosensitivity in glioma cells.

METHODS

The authors used glioma tissue samples from glioma patients, cells from glioblastoma (GBM) cell lines and normal human astrocyte cells, and subcutaneous tumor–bearing U87 cells in mice to examine the effects of signaling regulation by miR-448 in the response of glioma tissues and cells to radiation treatment. Techniques used for investigation included bioinformatics analyses, biochemical assays, luciferase reporter assays, and establishment of subcutaneous tumors in a mouse model. Glucose consumption, LDH activity, and cellular ATP were measured to determine the ability of glioma cells to perform glycolysis. Expression of HIF-1α was measured as a potential target gene of miR-448 in glycolysis.

RESULTS

miR-448 was detected and determined to be significantly downregulated in both glioma tissues from glioma patients and GBM cell lines. Furthermore, miR-448 acted as a tumor-inhibiting factor and suppressed glycolysis in glioma by negatively regulating the activity of HIF-1α signaling and then interfering with its downstream regulators relative to glycolysis, HK1, HK2, and LDHA. Interestingly, overexpression of miR-448 increased the x-radiation sensitivity of glioma cells. Finally, in in vivo experiments, subcutaneous tumor–bearing U87 cells in a mouse model verified that high expression of miR-448 also enhanced glioma radiosensitivity via inhibiting glycolytic factors.

CONCLUSIONS

miR-448 can promote radiosensitivity by inhibiting HIF-1α signaling and then negatively controlling the glycolysis process in glioma. A newly identified miR-448–HIF-1α axis acts as a potentially valuable therapeutic target that may be useful in overcoming radioresistance in glioma treatment.

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Timothee Jacquesson, Fang-Chang Yeh, Sandip Panesar, Jessica Barrios, Arnaud Attyé, Carole Frindel, Francois Cotton, Paul Gardner, Emmanuel Jouanneau and Juan C. Fernandez-Miranda

OBJECTIVE

Diffusion imaging tractography has allowed the in vivo description of brain white matter. One of its applications is preoperative planning for brain tumor resection. Due to a limited spatial and angular resolution, it is difficult for fiber tracking to delineate fiber crossing areas and small-scale structures, in particular brainstem tracts and cranial nerves. New methods are being developed but these involve extensive multistep tractography pipelines including the patient-specific design of multiple regions of interest (ROIs). The authors propose a new practical full tractography method that could be implemented in routine presurgical planning for skull base surgery.

METHODS

A Philips MRI machine provided diffusion-weighted and anatomical sequences for 2 healthy volunteers and 2 skull base tumor patients. Tractography of the full brainstem, the cerebellum, and cranial nerves was performed using the software DSI Studio, generalized-q-sampling reconstruction, orientation distribution function (ODF) of fibers, and a quantitative anisotropy–based generalized deterministic algorithm. No ROI or extensive manual filtering of spurious fibers was used. Tractography rendering was displayed in a tridimensional space with directional color code. This approach was also tested on diffusion data from the Human Connectome Project (HCP) database.

RESULTS

The brainstem, the cerebellum, and the cisternal segments of most cranial nerves were depicted in all participants. In cases of skull base tumors, the tridimensional rendering permitted the visualization of the whole anatomical environment and cranial nerve displacement, thus helping the surgical strategy.

CONCLUSIONS

As opposed to classical ROI-based methods, this novel full tractography approach could enable routine enhanced surgical planning or brain imaging for skull base tumors.

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Gabriel Crevier-Sorbo, Jeffrey Atkinson, Tanya Di Genova, Pramod Puligandla and Roy W. R. Dudley

Neurogenic stunned myocardium (NSM) is a potentially fatal cause of sudden cardiogenic dysfunction due to an acute neurological event, most commonly aneurysmal subarachnoid hemorrhage in adults. Only two pediatric cases of hydrocephalus-induced NSM have been reported. Here the authors report a third case in a 14-year-old boy who presented with severe headache, decreased level of consciousness, and shock in the context of acute hydrocephalus secondary to fourth ventricular outlet obstruction 3 years after standard-risk medulloblastoma treatment. He was initially stabilized with the insertion of an external ventricular drain and vasopressor treatment. He had a profoundly reduced cardiac contractility and became asystolic for 1 minute, requiring cardiopulmonary resuscitation when vasopressors were inadvertently discontinued. Over 1 week, his ventricles decreased in size and his cardiac function returned to normal. All other causes of heart failure were ruled out, and his impressive response to CSF diversion clarified the diagnosis of NSM secondary to hydrocephalus. He was unable to be weaned from his drain during his time in the hospital, so he underwent an endoscopic third ventriculostomy and has remained well with normal cardiac function at more than 6 months’ follow-up. This case highlights the importance of prompt CSF diversion and cardiac support for acute hydrocephalus presenting with heart failure in the pediatric population.

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Erlend Aambø Langvatn, Radek Frič, Bernt J. Due-Tønnessen and Per Kristian Eide

OBJECTIVE

Reduced intracranial volume (ICV) and raised intracranial pressure (ICP) are assumed to be principal pathophysiological mechanisms in childhood craniosynostosis. This study examined the association between ICV and ICP and whether ICV can be used to estimate the ICP.

METHODS

The authors analyzed ICV and ICP measurements from children with craniosynostosis without concurrent hydrocephalus and from age-matched individuals without craniosynostosis who underwent diagnostic ICP measurement.

RESULTS

The study included 19 children with craniosynostosis (mean age 2.2 ± 1.9 years) and 12 reference individuals without craniosynostosis (mean age 2.5 ± 1.6 years). There was no difference in ICV between the patient and reference cohorts. Both mean ICP (17.1 ± 5.6 mm Hg) and mean wave amplitude (5.9 ± 2.6 mm Hg) were higher in the patient cohort. The results disclosed no significant association between ICV and ICP values in the patient or reference cohorts, and no association was seen between change in ICV and ICP values after cranial vault expansion surgery (CVES) in 5 children in whom ICV and ICP were measured before and after CVES.

CONCLUSIONS

In this cohort of children with craniosynostosis, there was no significant association between ICV and ICP values prior to CVES and no significant association between change in ICV and ICP values after CVES in a subset of patients. Therefore, ICV could not reliably estimate the ICP values. The authors suggest that intracranial hypertension in childhood craniosynostosis may not be caused by reduced ICV alone but rather by a distorted relationship between ICV and the volume of intracranial content (brain tissue, CSF, and blood).