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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).

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Shahan Momjian, Rémi Tyrand, Basile N. Landis and Colette Boëx

OBJECTIVE

Intraoperative neuromonitoring of the chemical senses (smell and taste) has never been performed. The objective of this study was to determine if olfactory-evoked potentials could be obtained intraoperatively under general anesthesia.

METHODS

A standard olfactometer was used in the surgical theater with hydrogen sulfide (4 ppm, 200 msec). Olfactory-evoked potentials were recorded in 8 patients who underwent neurosurgery for resection of cerebral lesions. These patients underwent routine target-controlled propofol and sufentanil general anesthesia. Frontal, temporal, and parietal scalp subdermal electrodes were recorded ipsilaterally and contralaterally at the site of the surgery. Evoked potentials were computed if at least 70 epochs (0.5–100 Hz) satisfying the artifact rejection criterion (threshold 45 μV) could be extracted from signals of electrodes.

RESULTS

Contributive recordings were obtained for 5 of 8 patients (3 patients had fewer than 70 epochs with an amplitude < 45 μV). Olfactory-evoked potentials showed N1 responses (mean 442.8 ± 40.0 msec), most readily observed in the patient who underwent midline anterior fossa neurosurgery. No component of later latencies could be recorded consistently.

CONCLUSIONS

The study confirms that olfactory-evoked potentials can be measured in response to olfactory stimuli under general anesthesia. This demonstrates the feasibility of recording olfactory function intraoperatively and opens the potential for neuromonitoring of olfactory function during neurosurgery.

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Diana Ko, Daniel Blatt, Chafic Karam, Kunal Gupta and Ahmed M. Raslan

Nusinersen (Spinraza) is a US Food and Drug Administration–approved intrathecal medication for the treatment of spinal muscular atrophy (SMA). Adult patients with SMA often undergo thoracolumbar fusion to treat neurogenic scoliosis, preventing thecal access. The authors report a laminotomy technique and the ease of intrathecal access in three SMA patients with prior thoracolumbar fusions.

Patients were positioned in the lateral decubitus position or prone. Lumbar laminotomy was performed below the conus, between the lateral longitudinal rods, to preserve mechanical stability. Fluoroscopy provided real-time identification of instruments. Hardware was contoured with a carbide drill bit to develop the surgical window. Fiducial screws were placed along the perimeter for demarcation. Sublaminar wire removal caused dural defects that were repaired with a layer of dural substitute onlay and sealant. All patients successfully received nusinersen thecal injections via lumbar puncture by an interventional radiologist. Fluoroscopy time ranged from 6 to 36 seconds. No postoperative pseudomeningoceles, cerebrospinal fluid leaks, or wound complications occurred.

For patients with SMA and posterior fusion from prior scoliosis treatment, lumbar laminotomy is an effective method for creating thecal access for the administration of nusinersen.

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Zoe E. Teton, Daniel Blatt, Amr AlBakry, James Obayashi, Gulsah Ozturk, Vural Hamzaoglu, Philippe Magown, Nathan R. Selden, Kim J. Burchiel and Ahmed M. Raslan

OBJECTIVE

Despite rapid development and expansion of neuromodulation technologies, knowledge about device and/or therapy durability remains limited. The aim of this study was to evaluate the long-term rate of hardware and therapeutic failure of implanted devices for several neuromodulation therapies.

METHODS

The authors performed a retrospective analysis of patients’ device and therapy survival data (Kaplan-Meier survival analysis) for deep brain stimulation (DBS), vagus nerve stimulation (VNS), and spinal cord stimulation (SCS) at a single institution (years 1994–2015).

RESULTS

During the study period, 450 patients underwent DBS, 383 VNS, and 128 SCS. For DBS, the 5- and 10-year initial device survival was 87% and 73%, respectively, and therapy survival was 96% and 91%, respectively. For VNS, the 5- and 10-year initial device survival was 90% and 70%, respectively, and therapy survival was 99% and 97%, respectively. For SCS, the 5- and 10-year initial device survival was 50% and 34%, respectively, and therapy survival was 74% and 56%, respectively. The average initial device survival for DBS, VNS, and SCS was 14 years, 14 years, and 8 years while mean therapy survival was 18 years, 18 years, and 12.5 years, respectively.

CONCLUSIONS

The authors report, for the first time, comparative device and therapy survival rates out to 15 years for large cohorts of DBS, VNS, and SCS patients. Their results demonstrate higher device and therapy survival rates for DBS and VNS than for SCS. Hardware failures were more common among SCS patients, which may have played a role in the discontinuation of therapy. Higher therapy survival than device survival across all modalities indicates continued therapeutic benefit beyond initial device failures, which is important to emphasize when counseling patients.

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Henrik Giese, Benjamin Haenig, Anna Haenig, Andreas Unterberg and Klaus Zweckberger

OBJECTIVE

Craniopharyngiomas are rare and benign tumors of the sellar and/or parasellar region. Primary treatment involves resection followed by adjuvant radiotherapy. While the grade of resection was frequently analyzed following surgery, the neurological outcome and especially neuropsychological deficits and quality of life have been neglected for many decades. Therefore, the authors retrospectively analyzed their patient series and prospectively assessed neuropsychological outcome and quality of life following resection of craniopharyngiomas in adults.

METHODS

In total, 71 patients (39 men and 32 women) with a mean age of 49 years were enrolled in the retrospective analysis. In addition, 36 of the 71 patients were included in the prospective arm of the study and underwent neurological and neuropsychological testing as well as quality of life (36-Item Short-Form Health Survey; SF-36) assessment. Factors influencing outcome were identified and correlations calculated.

RESULTS

Resection was performed mostly using a pterional (41.6%, 47/113 surgical procedures) or bifrontal translamina terminalis (30.1%, 34/113 surgical procedures) approach. Following surgery, visual acuity was significantly improved (> 0.2 diopters) in 32.4% (23/71) of patients, or remained stable in 45.1% (32/71) of patients. During long-term follow up, 80.3% (57/71) of patients developed pituitary insufficiency, particularly involving the corticotropic and thyrotrophic axes. In total, 75% (27/36) of patients showed neuropsychological deviations in at least 1 test item. In particular, attentiveness, cognitive speed, and short-term memory were affected. Referring to the SF-36 score, quality of life was affected in both the mental and physical score in 19.4% (7/36) and 33.3% (12/36), respectively. The risk factors that were identified were a tumor volume larger than 9 cm3, tumor extension toward/into the third ventricle or the brainstem, and resection using a bifrontal translamina terminalis or left-sided approach.

CONCLUSIONS

This study demonstrated that resection of craniopharyngiomas is frequently associated with postoperative neuropsychological deficits and hence an impaired quality of life. In addition to tumor size and extension toward/into the third ventricle or the brainstem, selection of the surgical approach may play a crucial role in the patient’s neuropsychological outcome and quality of life.

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Tonje Haug Nordenmark, Tanja Karic, Cecilie Røe, Wilhelm Sorteberg and Angelika Sorteberg

OBJECTIVE

Although many patients recover to a good functional outcome after aneurysmal subarachnoid hemorrhage (aSAH), residual symptoms are very common and may have a large impact on the patient’s daily life. The particular cluster of residual symptoms after aSAH has not previously been described in detail and there is no validated questionnaire that covers the typical problems reported after aSAH. Many of the symptoms are similar to post-concussion syndrome, which often is evaluated with the Rivermead Post-Concussion Symptoms Questionnaire (RPQ). In the present study, the authors therefore performed an exploratory use of the RPQ as a template to describe post-aSAH syndrome.

METHODS

The RPQ was administered to 128 patients in the chronic phase after aSAH along with a battery of quality-of-life questionnaires. The patients also underwent a medical examination besides cognitive and physical testing. Based on their RPQ scores, patients were dichotomized into a “syndrome” group or “recovery” group.

RESULTS

A post-aSAH syndrome was seen in 33% of the patients and their symptom burden on all RPQ subscales was significantly higher than that of patients who had recovered on all RPQ subscales. The symptom cluster consisted mainly of fatigue, cognitive problems, and emotional problems. Physical problems were less frequently reported. Patients with post-aSAH syndrome scored significantly worse on mobility and pain scores, as well as on quality-of-life questionnaires. They also had significantly poorer scores on neuropsychological tests of verbal learning, verbal short- and long-term memory, psychomotor speed, and executive functions. Whereas 36% of the patients in the recovery group were able to return to their premorbid occupational status, this was true for only 1 patient in the syndrome group.

CONCLUSIONS

Approximately one-third of aSAH patients develop a post-aSAH syndrome. These patients struggle with fatigue and cognitive and emotional problems. Patients with post-aSAH syndrome report more pain and reduced quality of life compared to patients without this cluster of residual symptoms and have larger cognitive deficits. In this sample, patients with post-aSAH syndrome were almost invariably excluded from return to work. The RPQ is a simple questionnaire covering the specter of residual symptoms after aSAH. Being able to acknowledge these patients’ complaints as a defined syndrome using the RPQ should help patients to accept and cope, thereby alleviating possible secondary distress produced.

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Andrew T. Hale, Amanda N. Stanton, Shilin Zhao, Faizal Haji, Stephen R. Gannon, Anastasia Arynchyna, John C. Wellons, Brandon G. Rocque and Robert P. Naftel

OBJECTIVE

At failure of endoscopic third ventriculostomy (ETV) with choroid plexus cauterization (CPC), the ETV ostomy may be found to be closed or open. Failure with a closed ostomy may indicate a population that could benefit from evolving techniques to keep the ostomy open and may be candidates for repeat ETV, whereas failure with an open ostomy may be due to persistently abnormal CSF dynamics. This study seeks to identify clinical and radiographic predictors of ostomy status at the time of ETV/CPC failure.

METHODS

The authors conducted a multicenter, retrospective cohort study on all pediatric patients with hydrocephalus who failed initial ETV/CPC treatment between January 2013 and October 2016. Failure was defined as the need for repeat ETV or ventriculoperitoneal (VP) shunt placement. Clinical and radiographic data were collected, and ETV ostomy status was determined endoscopically at the subsequent hydrocephalus procedure. Statistical analysis included the Mann-Whitney U-test, Wilcoxon rank-sum test, t-test, and Pearson chi-square test where appropriate, as well as multivariate logistic regression.

RESULTS

Of 72 ETV/CPC failures, 28 patients (39%) had open-ostomy failure and 44 (61%) had closed-ostomy failure. Patients with open-ostomy failure were older (median 5.1 weeks corrected age for gestation [interquartile range (IQR) 0.9–15.9 weeks]) than patients with closed-ostomy failure (median 0.2 weeks [IQR −1.3 to 4.5 weeks]), a significant difference by univariate and multivariate regression. Etiologies of hydrocephalus included intraventricular hemorrhage of prematurity (32%), myelomeningocele (29%), congenital communicating (11%), aqueductal stenosis (11%), cyst/tumor (4%), and other causes (12%). A wider baseline third ventricle was associated with open-ostomy failure (median 15.0 mm [IQR 10.3–18.5 mm]) compared to closed-ostomy failure (median 11.7 mm [IQR 8.9–16.5 mm], p = 0.048). Finally, at the time of failure, patients with closed-ostomy failure had enlargement of their ventricles (frontal and occipital horn ratio [FOHR], failure vs baseline, median 0.06 [IQR 0.00–0.11]), while patients with open-ostomy failure had no change in ventricle size (median 0.01 [IQR −0.04 to 0.05], p = 0.018). Previous CSF temporizing procedures, intraoperative bleeding, and time to failure were not associated with ostomy status at ETV/CPC failure.

CONCLUSIONS

Older corrected age for gestation, larger baseline third ventricle width, and no change in FOHR were associated with open-ostomy ETV/CPC failure. Future studies are warranted to further define and confirm features that may be predictive of ostomy status at the time of ETV/CPC failure.

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Joshua L. Golubovsky, Arbaz Momin, Nicolas R. Thompson and Michael P. Steinmetz

OBJECTIVE

Bertolotti syndrome is a rare spinal condition that causes low-back pain due to a lumbosacral transitional vertebra (LSTV), which is a pseudoarticulation between the fifth lumbar transverse process and the sacral ala. Bertolotti syndrome patients are rarely studied, particularly with regard to their quality of life. This study aimed to examine the quality of life and prior treatments in patients with Bertolotti syndrome at first presentation to the authors’ center in comparison with those with lumbosacral radiculopathy.

METHODS

This study was a retrospective cohort analysis of patients with Bertolotti syndrome and lumbosacral radiculopathy due to disc herniation seen at the authors’ institution’s spine center from 2005 through 2018. Diagnoses were confirmed with provider notes and imaging. Variables collected included demographics, diagnostic history, prior treatment, patient-reported quality of life metrics, and whether or not they underwent surgery at the authors’ institution. Propensity score matching by age and sex was used to match lumbosacral radiculopathy patients to Bertolotti syndrome patients. Group comparisons were made using t-tests, Fisher’s exact test, Mann-Whitney U-tests, Cox proportional hazards models, and linear regression models where variables found to be different at the univariate level were included as covariates.

RESULTS

The final cohort included 22 patients with Bertolotti syndrome who had patient-reported outcomes data available and 46 propensity score–matched patients who had confirmed radiculopathy due to disc herniation. The authors found that Bertolotti syndrome patients had significantly more prior epidural steroid injections (ESIs) and a longer time from symptom onset to their first visit. Univariate analysis showed that Bertolotti syndrome patients had significantly worse Patient-Reported Outcomes Measurement Information System (PROMIS) mental health T-scores. Adjustment for prior ESIs and time from symptom onset revealed that Bertolotti syndrome patients also had significantly worse PROMIS physical health T-scores. Time to surgery and other quality of life metrics did not differ between groups.

CONCLUSIONS

Patients with Bertolotti syndrome undergo significantly longer workup and more ESIs and have worse physical and mental health scores than age- and sex-matched patients with lumbosacral radiculopathy. However, both groups of patients had mild depression and clinically meaningful reduction in their quality of life according to all instruments. This study shows that Bertolotti syndrome patients have a condition that affects them potentially more significantly than those with lumbosacral radiculopathy, and increased attention should be paid to these patients to improve their workup, diagnosis, and treatment.