Publications by authors named "Mark Luciano"

73 Publications

Cognitive and gait outcomes after primary endoscopic third ventriculostomy in adults with chronic obstructive hydrocephalus.

J Neurosurg 2021 Sep 17:1-8. Epub 2021 Sep 17.

12Department of Clinical Neurosciences, Division of Neurosurgery, University of Calgary Cumming School of Medicine, Calgary, Alberta, Canada.

Objective: The object of this study was to determine the short- and long-term efficacy of primary endoscopic third ventriculostomy (ETV) on cognition and gait in adults with chronic obstructive hydrocephalus.

Methods: Patients were prospectively accrued through the Adult Hydrocephalus Clinical Research Network patient registry. Patients with previously untreated congenital or acquired obstructive hydrocephalus were included in this study. Gait velocity was assessed using a 10-m walk test. Global cognition was assessed with the Montreal Cognitive Assessment (MoCA). Only patients with documented pre- and post-ETV gait analysis and/or pre- and post-ETV MoCA were included.

Results: A total of 74 patients had undergone primary ETV, 42 of whom were analyzed. The remaining 32 patients were excluded, as they could not complete both pre- and post-ETV assessments. The mean age of the 42 patients, 19 (45.2%) of whom were female, was 51.9 ± 17.1 years (range 19-79 years). Most patients were White (37 [88.1%]), and the remainder were Asian. Surgical complications were minor. Congenital etiologies occurred in 31 patients (73.8%), with aqueductal stenosis in 23 of those patients (54.8%). The remaining 11 patients (26.2%) had acquired cases. The gait short-term follow-up cohort (mean 4.7 ± 4.1 months, 35 patients) had a baseline median gait velocity of 0.9 m/sec (IQR 0.7-1.3 m/sec) and a post-ETV median velocity of 1.3 m/sec (IQR 1.1-1.4 m/sec). Gait velocity significantly improved post-ETV with a median within-patient change of 0.3 m/sec (IQR 0.0-0.6 m/sec, p < 0.001). Gait velocity improvements were sustained in the long term (mean 14 ± 2.8 months, 12 patients) with a baseline median velocity of 0.7 m/sec (IQR 0.6-1.3 m/sec), post-ETV median of 1.3 m/sec (IQR 1.1-1.7 m/sec), and median within-patient change of 0.4 m/sec (IQR 0.2-0.6 m/sec, p < 0.001). The cognitive short-term follow-up cohort (mean 4.6 ± 4.0 months, 38 patients) had a baseline median MoCA total score (MoCA TS) of 24/30 (IQR 23-27) that improved to 26/30 (IQR 24-28) post-ETV. The median within-patient change was +1 point (IQR 0-2 points, p < 0.001). However, this change is not clinically significant. The cognitive long-term follow-up cohort (mean 14 ± 3.1 months, 15 patients) had a baseline median MoCA TS of 23/30 (IQR 22-27), which improved to 26/30 (IQR 25-28) post-ETV. The median within-patient change was +2 points (IQR 1-3 points, p = 0.007), which is both statistically and clinically significant.

Conclusions: Primary ETV can safely improve symptoms of gait and cognitive dysfunction in adults with chronic obstructive hydrocephalus. Gait velocity and global cognition were significantly improved, and the worsening of either was rare following ETV.
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http://dx.doi.org/10.3171/2021.3.JNS203424DOI Listing
September 2021

Intraoperative cone-beam and slot-beam CT: 3D image quality and dose with a slot collimator on the O-arm imaging system.

Med Phys 2021 Sep 14. Epub 2021 Sep 14.

Department of Biomedical Engineering, Johns Hopkins University, Baltimore, MD, USA.

Purpose: To characterize the 3D imaging performance and radiation dose for a prototype slot-beam configuration on an intraoperative O-arm™ Surgical Imaging System (Medtronic Inc., Littleton MA) and identify potential improvements in soft-tissue image quality for surgical interventions.

Methods: A slot collimator was integrated with the O-arm system for slot-beam axial CT. The collimator can be automatically actuated to provide 1.2° slot-beam longitudinal collimation. Cone-beam and slot-beam configurations were investigated with and without an antiscatter grid (12:1 grid ratio, 60 lines/cm). Dose, scatter, image noise, and soft-tissue contrast resolution were evaluated in quantitative phantoms for head and body configurations over a range of exposure levels (beam energy and mAs), with reconstruction performed via filtered-backprojection. Qualitative imaging performance across various anatomical sites and imaging tasks was assessed with anthropomorphic head, abdomen, and pelvis phantoms.

Results: The dose for a slot-beam scan varied from 0.02-0.06 mGy/mAs for head protocols to 0.01-0.03 mGy/mAs for body protocols, yielding dose reduction by ∼1/5 to 1/3 compared to cone-beam, owing to beam collimation and reduced x-ray scatter. The slot-beam provided a ∼6-7× reduction in scatter-to-primary ratio (SPR) compared to the cone-beam, yielding SPR ∼20%-80% for head and body without the grid and ∼7%-30% with the grid. Compared to cone-beam scans at equivalent dose, slot-beam images exhibited a ∼2.5× increase in soft-tissue CNR for both grid and gridless configurations. For slot-beam scans, a further ∼10-30% improvement in CNR was achieved when the grid was removed. Slot-beam imaging could benefit certain interventional scenarios in which improved visualization of soft tissues is required within a fairly narrow longitudinal region of interest (7 mm in ) - e.g., checking the completeness of tumor resection, preservation of adjacent anatomy, or detection of complications (e.g., hemorrhage). While preserving existing capabilities for fluoroscopy and cone-beam CT, slot-beam scanning could enhance the utility of intraoperative imaging and provide a useful mode for safety and validation checks in image-guided surgery.

Conclusions: The 3D imaging performance and dose of a prototype slot-beam CT configuration on the O-arm system was investigated. Substantial improvements in soft-tissue image quality and reduction in radiation dose are evident with the slot-beam configuration due to reduced x-ray scatter. This article is protected by copyright. All rights reserved.
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http://dx.doi.org/10.1002/mp.15221DOI Listing
September 2021

The Impact of Hydrocephalus Shunt Devices on Quality of Life.

J Craniofac Surg 2021 Jul-Aug 01;32(5):1746-1750

Section of Neuroplastic and Reconstructive Surgery, Department of Plastic and Reconstructive Surgery.

Background: Despite advances in hydrocephalus shunt technology and improvement in hydrocephalus management, many patients have chronic disability and require multiple surgeries throughout their lifetime. There is limited data from patients' perspective regarding the impact of shunt devices on quality-of-life.

Methods: A cross-sectional survey was developed to evaluate the impact of shunt devices on patient quality-of-life. The survey was distributed via social media platforms of the Hydrocephalus Association, and patients self-selected to anonymously complete the online questionnaire. A literature review was performed to contextualize the findings from the survey.

Results: A total of 562 survey responses were obtained from a network encompassing 35,000 members. The mean age was 30 years old (0.5-87), and 65% identified as female. Eighty one percent underwent at least 1 shunt revision surgery, with a reported average of 10 shunt revision surgeries per patient (1-200 surgeries). Occlusion, shunt migration and infection were the leading causes for revision at 60%, 47%, and 35%, respectively. In addition, 72% of patients reported pain and discomfort from the device, and 68% expressed avoidance of certain activities due to "fear of bumping shunt." Despite numerous articles discussing shunt technology, a review of the literature indicated a paucity of studies specifically evaluating the burden of shunt devices from a patient/caregiver perspective.

Conclusions: The findings from this study suggest long-term physical and psychosocial burden associated with shunt devices. Importantly, this study highlights the need for concerted efforts to develop validated tools to study patient reported outcomes as it relates to neurocranial implanted devices.
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http://dx.doi.org/10.1097/SCS.0000000000007579DOI Listing
July 2021

Impact of international research fellows in neurosurgery: results from a single academic center.

J Neurosurg 2021 Jul 23:1-11. Epub 2021 Jul 23.

Objective: International research fellows have been historically involved in academic neurosurgery in the United States (US). To date, the contribution of international research fellows has been underreported. Herein, the authors aimed to quantify the academic output of international research fellows in the Department of Neurosurgery at The Johns Hopkins University School of Medicine.

Methods: Research fellows with Doctor of Medicine (MD), Doctor of Philosophy (PhD), or MD/PhD degrees from a non-US institution who worked in the Hopkins Department of Neurosurgery for at least 6 months over the past decade (2010-2020) were included in this study. Publications produced during fellowship, number of citations, and journal impact factors (IFs) were analyzed using ANOVA. A survey was sent to collect information on personal background, demographics, and academic activities.

Results: Sixty-four international research fellows were included, with 42 (65.6%) having MD degrees, 17 (26.6%) having PhD degrees, and 5 (7.8%) having MD/PhD degrees. During an average 27.9 months of fellowship, 460 publications were produced in 136 unique journals, with 8628 citations and a cumulative journal IF of 1665.73. There was no significant difference in total number of publications, first-author publications, and total citations per person among the different degree holders. Persons holding MD/PhDs had a higher number of citations per publication per person (p = 0.027), whereas those with MDs had higher total IFs per person (p = 0.048). Among the 43 (67.2%) survey responders, 34 (79.1%) had nonimmigrant visas at the start of the fellowship, 16 (37.2%) were self-paid or funded by their country of origin, and 35 (81.4%) had mentored at least one US medical student, nonmedical graduate student, or undergraduate student.

Conclusions: International research fellows at the authors' institution have contributed significantly to academic neurosurgery. Although they have faced major challenges like maintaining nonimmigrant visas, negotiating cultural/language differences, and managing self-sustainability, their scientific productivity has been substantial. Additionally, the majority of fellows have provided reciprocal mentorship to US students.
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http://dx.doi.org/10.3171/2021.1.JNS203824DOI Listing
July 2021

Evaluation of the effect comorbid Parkinson syndrome on normal pressure hydrocephalus assessment.

Clin Neurol Neurosurg 2021 08 10;207:106810. Epub 2021 Jul 10.

Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD, USA; Department of Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, USA.

Objective: The primary aim of the study was to assess the effect comorbid Parkinson syndromes have on results of CSF tap test (TT) and shunt outcomes for patients presenting with Normal Pressure Hydrocephalus (NPH). We hypothesized that patients with possible NPH and comorbid Parkinson syndromes with Positive DaT scans will not respond to CSF TT at the same rate as patients without comorbid Parkinson syndromes. Additionally, we followed a small number of patients with positive DaT scans who were shunted to assess long term outcome of comorbid Parkinson syndromes.

Methods: Medical records and neurological exams of 251 patients were reviewed. In our analysis 101 patients with no parkinsonian symptoms and no DaT scans were included as a control group, there were 52 patients with DaT scans, 31 patients were positive (DaT-P). Gait measures were assessed before and after CSF TT using the Wilcoxon matched-pairs signed-rank test or paired t-tests were used. To compare the effect of DaT-P and Control, we used an ANCOVA controlling for age, sex, assistive device used, and past medical history effecting gait.

Results: There was not a significant difference in response between Control and DaT-P group. The Control group improved on timed up and go (TUG) by 14.82%, DualTUG 16.35%, 10-meter Walk Test (10MWT) 18.13%, MiniBEST 15.91%, and 6-minute Walk Test (6MWT) 13.96%, while the DaT-P group improved on TUG by 14.93%, DualTUG 17.24%, 10MWT 22.68%, MiniBEST 18.07%, and 6MWT 16.06%.

Conclusion: Our findings suggest that patients with possible NPH and suspected comorbid movement disorder, showed similar improvement after diagnostic CSF TT compared to participants with no parkinsonian symptoms present on exam.

Data Availability Statement: Data relevant to the study will be made available from the corresponding author upon a reasonable request.
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http://dx.doi.org/10.1016/j.clineuro.2021.106810DOI Listing
August 2021

Functional connectivity abnormalities in Type I Chiari: associations with cognition and pain.

Brain Commun 2021 Jul 14;3(3):fcab137. Epub 2021 Jun 14.

Department of Psychology, The University of Akron, Akron, OH 44325, USA.

There is initial evidence of microstructural abnormalities in the fibre-tract pathways of the cerebellum and cerebrum of individuals diagnosed with Type I Chiari malformation. However, it is unclear whether abnormal white matter architecture and macro-level morphological deviations that have been observed in Chiari translate to differences in functional connectivity. Furthermore, common symptoms of Chiari include pain and cognitive deficits, but the relationship between these symptoms and functional connectivity has not been explored in this population. Eighteen Type I Chiari patients and 18 age-, sex- and education-matched controls underwent resting-state functional MRI to measure functional connectivity. Participants also completed a neuropsychological battery and completed self-report measures of chronic pain. Group differences in functional connectivity were identified. Subsequently, pathways of significant difference were re-analyzed after controlling for the effects of attention performance and self-reported chronic pain. Chiari patients exhibited functional hypoconnectivity between areas of the cerebellum and cerebrum. Controlling for attention eliminated all deficits with the exception of that from the posterior cerebellar pathway. Similarly, controlling for pain also eliminated deficits except for those from the posterior cerebellar pathway and vermis VII. Patterns of Chiari hyperconnectivity were also found between regions of the cerebellum and cerebrum in Chiari patients. Hyperconnectivity in all regions was eliminated after controlling for attention except between left lobule VIII and the left postcentral gyrus and between vermis IX and the precuneus. Similarly, hyperconnectivity was eliminated after controlling for pain except between the default mode network and globus pallidus, left lobule VIII and the left postcentral gyrus, and Vermis IX and the precuneus. Evidence of both hyper- and hypoconnectivity were identified in Chiari, which is posited to support the hypothesis that the effect of increased pain in Chiari draws on neural resources, requiring an upregulation in inhibitory control mechanisms and resulting in cognitive dysfunction. Areas of hypoconnectivity in Chiari patients also suggest disruption in functional pathways, and potential mechanisms are discussed.
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http://dx.doi.org/10.1093/braincomms/fcab137DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8279071PMC
July 2021

A proposed framework for cerebral venous congestion.

Neuroradiol J 2021 Jul 5:19714009211029261. Epub 2021 Jul 5.

Department of Radiology and Radiological Science, Johns Hopkins University School of Medicine, USA.

Background: While venous congestion in the peripheral vasculature has been described and accepted, intracranial venous congestion remains poorly understood. The characteristics, pathophysiology, and management of cerebral venous stasis, venous hypertension and venous congestion remain controversial, and a unifying conceptual schema is absent. The cerebral venous and lymphatic systems are part of a complex and dynamic interaction between the intracranial compartments, with interplay between the parenchyma, veins, arteries, cerebrospinal fluid, and recently characterized lymphatic-like systems in the brain. Each component contributes towards intracranial pressure, occupying space within the fixed calvarial volume. This article proposes a framework to consider conditions resulting in brain and neck venous congestion, and seeks to expedite further study of cerebral venous diagnoses, mechanisms, symptomatology, and treatments.

Methods: A multi-institution retrospective review was performed to identify unique patient cases, complemented with a published case series to assess a spectrum of disease states with components of venous congestion affecting the brain. These diseases were organized according to anatomical location and purported mechanisms. Outcomes of treatments were also analyzed. Illustrative cases were identified in the venous treatment databases of the authors.

Conclusion: This framework is the first clinically structured description of venous pathologies resulting in intracranial venous and cerebrospinal fluid hypertension. Our proposed system highlights unique clinical symptoms and features critical for appropriate diagnostic work-up and potential treatment. This novel schema allows clinicians effectively to approach cases of intracranial hypertension secondary to venous etiologies, and furthermore provides a framework by which researchers can better understand this developing area of cerebrovascular disease.
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http://dx.doi.org/10.1177/19714009211029261DOI Listing
July 2021

Adult Age Differences in Self-Reported Pain and Anterior CSF Space in Chiari Malformation.

Cerebellum 2021 Jun 9. Epub 2021 Jun 9.

Department of Psychology, The University of Akron, Akron, OH, 44325-4301, USA.

Chiari malformation type I (CMI) is a neural disorder with sensory, cognitive, and motor defects, as well as headaches. Radiologically, the cerebellar tonsils extend below the foramen magnum. To date, the relationships among adult age, brain morphometry, surgical status, and symptom severity in CMI are unknown. The objective of this study was to better understand the relationships among these variables using causal modeling techniques. Adult CMI patients (80% female) who either had (n = 150) or had not (n = 151) undergone posterior fossa decompression surgery were assessed using morphometric measures derived from magnetic resonance images (MRI). MRI-based morphometry showed that the area of the CSF pocket anterior to the cervico-medullary junction (anterior CSF space) correlated with age at the time of MRI (r =  - .21). Also, self-reported pain increased with age (r = .11) and decreased with anterior CSF space (r =  - .18). Age differences in self-reported pain were mediated by anterior CSF space in the cervical spine area-and this effect was particularly salient for non-decompressed CMI patients. As CMI patients age, the anterior CSF space decreases, and this is associated with increased pain-especially for non-decompressed CMI patients. It is recommended that further consideration of age-related decreases in anterior CSF space in CMI patients be given in future research.
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http://dx.doi.org/10.1007/s12311-021-01289-wDOI Listing
June 2021

In vitro evaluation of cerebrospinal fluid velocity measurement in type I Chiari malformation: repeatability, reproducibility, and agreement using 2D phase contrast and 4D flow MRI.

Fluids Barriers CNS 2021 Mar 18;18(1):12. Epub 2021 Mar 18.

Department of Chemical and Biological Engineering, University of Idaho, 875 Perimeter Dr. MC1122, Moscow, ID, 83844, USA.

Background: Phase contrast magnetic resonance imaging, PC MRI, is a valuable tool allowing for non-invasive quantification of CSF dynamics, but has lacked adoption in clinical practice for Chiari malformation diagnostics. To improve these diagnostic practices, a better understanding of PC MRI based measurement agreement, repeatability, and reproducibility of CSF dynamics is needed.

Methods: An anatomically realistic in vitro subject specific model of a Chiari malformation patient was scanned three times at five different scanning centers using 2D PC MRI and 4D Flow techniques to quantify intra-scanner repeatability, inter-scanner reproducibility, and agreement between imaging modalities. Peak systolic CSF velocities were measured at nine axial planes using 2D PC MRI, which were then compared to 4D Flow peak systolic velocity measurements extracted at those exact axial positions along the model.

Results: Comparison of measurement results showed good overall agreement of CSF velocity detection between 2D PC MRI and 4D Flow (p = 0.86), fair intra-scanner repeatability (confidence intervals ± 1.5 cm/s), and poor inter-scanner reproducibility. On average, 4D Flow measurements had a larger variability than 2D PC MRI measurements (standard deviations 1.83 and 1.04 cm/s, respectively).

Conclusion: Agreement, repeatability, and reproducibility of 2D PC MRI and 4D Flow detection of peak CSF velocities was quantified using a patient-specific in vitro model of Chiari malformation. In combination, the greatest factor leading to measurement inconsistency was determined to be a lack of reproducibility between different MRI centers. Overall, these findings may help lead to better understanding for application of 2D PC MRI and 4D Flow techniques as diagnostic tools for CSF dynamics quantification in Chiari malformation and related diseases.
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http://dx.doi.org/10.1186/s12987-021-00246-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7977612PMC
March 2021

Impact of Surgical Status, Loneliness, and Disability on Interleukin 6, C-Reactive Protein, Cortisol, and Estrogen in Females with Symptomatic Type I Chiari Malformation.

Cerebellum 2021 Mar 6. Epub 2021 Mar 6.

Department of Neurosurgery, Johns Hopkins Medical Center, Baltimore, MD, USA.

Chiari malformation type I (CMI) provides an opportunity for examining possible moderators of allostatic load. CMI patients who had (n = 43) and had not (n = 19) undergone decompression surgery completed questionnaires regarding pain, disability, and loneliness, and provided serum samples for IL-6, CRP, estrogen, and free estradiol assays, and saliva samples to assess diurnal cortisol curves. ANOVAs examining surgical status (decompressed versus non-decompressed), loneliness (high vs. low), and disability (high vs. low) as independent variables and biomarker variables as dependent factors found that loneliness was associated with higher levels of cortisol, F(1, 37) = 4.91, p = .04, η = .11, and lower levels of estrogen, F(1, 36) = 7.29, p = .01, η = .17, but only in decompressed patients. Results highlight the possible impact of loneliness on biological stress responses and the need to intervene to reduce loneliness in patients with symptomatic CMI.
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http://dx.doi.org/10.1007/s12311-021-01251-wDOI Listing
March 2021

Assessing the predictive value of common gait measure for predicting falls in patients presenting with suspected normal pressure hydrocephalus.

BMC Neurol 2021 Feb 8;21(1):60. Epub 2021 Feb 8.

Department of Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, USA.

Objective: To assess the predictive value of common measures validated to predict falls in other geriatric populations in patients presenting with suspected Normal Pressure Hydrocephalus (NPH).

Methods: One hundred ninety-five patients over the age of 60 who received the Fall Risk Questionnaire were retrospectively recruited from the CSF Disorders clinic within the departments of Neurosurgery and Neurology. Multiple logistic regression was used to create a model to predict falls for patients with suspected NPH using common measures: Timed Up & Go, Dual Timed Up & Go, 10 Meter Walk, MiniBESTest, 6-Minute Walk, Lower Extremity Function (Mobility), Fall Risk Questionnaire, and Functional Activities Questionnaire.

Results: The Fall Risk Questionnaire and age were shown to be the best predictors of falls. The model was 95.92% (Positive predictive value: 83.93%) sensitive and 47.92% specific (Negative predictive value: 77.78%).

Conclusion: Patients presenting with suspected NPH are at an increased fall risk, 75% of the total patients and 89% of patients who responded to a temporary drain of CSF had at least one fall in the past 6 months. The Fall Risk Questionnaire and age were shown to be predictive of falls for patients with suspected NPH. The preliminary evidence indicates measures that have been validated to assess fall risk in other populations may not be valid for patients presenting with suspected NPH.
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http://dx.doi.org/10.1186/s12883-021-02068-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7869204PMC
February 2021

Investigation of a Valve-Agnostic Cranial Implant for Adult Hydrocephalus Patients Requiring Ventriculoperitoneal Shunting.

J Craniofac Surg 2020 Oct;31(7):1998-2002

Section of Neuroplastic and Reconstructive Surgery, Departments of Plastic Surgery and Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, MD.

Introduction: Currently, the most effective treatment strategy for adults with hydrocephalus involves cerebrospinal fluid diversion by means of a shunt system, most commonly ventriculoperitoneal shunts (VPS). Ventriculoperitoneal shunting is associated with high complication and/or revision rates, in part due to the high-profile programmable valve designs. Thus, the valve-agnostic cranial implant (VACI) was designed and investigated as a safe and effective method of reducing the valve's high profile and is currently undergoing clinical trials. As such, the objective of this study was to collate preliminary, multi-institutional data of early outcomes using a VACI approach for patients requiring VPS by way of an Institutional Review Board approved registry.

Methods: A total of 25 adult patients across 4 institutions and 6 surgeons underwent VACI placement for VPS based on preoperative evaluation and perceived benefit. Patient demographics, operative details, and preliminary outcomes are presented here.

Results: Valve-agnostic cranial implant placement via a limited size craniectomy at time of shunt revision was performed with no adverse events. Over an average follow-up period of 1 year (394 ± 178 days), 92% of patients experienced no major shunt-related or scalp-related complications. There were 2 cases with a major complication requiring reoperation: 1 shunt tubing extrusion and 1 case of meningitis. The most frequent postsurgical intervention seen in this study was related to adjustment of drainage: a non-invasively performed valve reprogramming after initial shunt placement when proper flow rate is being established. Of the 8 cases of drainage adjustment, all but 1 (88%) were receiving a VPS for the first time, with the exception undergoing a fourth shunt revision. All instances of improper flow were treated non-surgically and remediated effectively via shunt reprogramming in clinic. Removal of the VACI was not indicated in any treatment course. In this way, all complications as they relate to the shunt valve were minor and required nonsurgical intervention, and no complications reported were directly or indirectly caused by using the VACI.

Conclusion: Preliminary findings from this multicenter trial suggest promising outcomes with a low complication rate for patients with hydrocephalus undergoing VACI placement during VPS. Ongoing research will continue to provide a more robust clinical picture of VACI in hydrocephalus management as more data becomes available.
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http://dx.doi.org/10.1097/SCS.0000000000006730DOI Listing
October 2020

Extended lumbar drainage in idiopathic normal pressure hydrocephalus: a systematic review and meta-analysis of diagnostic test accuracy.

Br J Neurosurg 2021 Jun 9;35(3):285-291. Epub 2020 Jul 9.

Department of Neurosurgery, Southmead Hospital, Bristol, UK.

Background: When appropriately selected, a high proportion of patients with suspected idiopathic normal pressure hydrocephalus (iNPH) will respond to cerebrospinal fluid diversion with a shunt. Extended lumbar drainage (ELD) is regarded as the most accurate test for this condition, however, varying estimates of its accuracy are found in the current literature. Here, we review the literature in order to provide summary estimates of sensitivity, specificity, positive- and negative predictive value for this test through meta-analysis of suitably rigorous studies.

Methods: Studies involving a population of NPH patients with predominantly idiopathic aetiology (>80%) in which the intention of the study was to shunt patients regardless of the outcome of ELD were included in the review. Various literature databases were searched to identify diagnostic test accuracy studies addressing ELD in the diagnosis of iNPH. Those studies passing screening and eligibility were assessed using the QUADAS-2 tool and data extracted for bivariate random effects meta-analysis.

Results: Four small studies were identified. They showed disparate results concerning diagnostic test accuracy. The summary estimates for sensitivity and specificity were 94% (CI 41-100%) and 85% (CI 33-100%), respectively. The summary estimates of positive and negative predictive value were both 90% (CIs 65-100% and 48-100%, respectively).

Conclusion: Large, rigorous studies addressing the diagnostic accuracy of ELD are lacking, and little robust evidence exists to support the use of ELD in diagnostic algorithms for iNPH. Therefore, a large cohort study, or ideally an RCT, is needed to determine best practice in selecting patients for shunt surgery.
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http://dx.doi.org/10.1080/02688697.2020.1787948DOI Listing
June 2021

Minimally invasive therapeutic ultrasound: Ultrasound-guided ultrasound ablation in neuro-oncology.

Ultrasonics 2020 Dec 20;108:106210. Epub 2020 Jun 20.

Department of Biomedical Engineering, Johns Hopkins University, Baltimore, MD, USA; Department of Neurosurgery, Johns Hopkins University, Baltimore, MD, USA. Electronic address:

Introduction: To improve patient outcomes (eg, reducing blood loss and infection), practitioners have gravitated toward noninvasive and minimally invasive surgeries (MIS), which demand specialized toolkits. Focused ultrasound, for example, facilitates thermal ablation from a distance, thereby reducing injury to surrounding tissue. Focused ultrasound can often be performed noninvasively; however, it is more difficult to carry out in neuro-oncological tumors, as ultrasound is dramatically attenuated while propagating through the skull. This shortcoming has prompted exploration of MIS options for intracranial placement of focused ultrasound probes, such as within the BrainPath™ (NICO Corporation, Indianapolis, IN). Herein, we present the design, development, and in vitro testing of an image-guided, focused ultrasound prototype designed for use in MIS procedures. This probe can ablate neuro-oncological lesions despite its small size.

Materials & Methods: Preliminary prototypes were iteratively designed, built, and tested. The final prototype consisted of three 8-mm-diameter therapeutic elements guided by an imaging probe. Probe functionality was validated on a series of tissue-mimicking phantoms.

Results: Lesions were created in tissue-mimicking phantoms with average dimensions of 2.5 × 1.2 × 6.5 mm and 3.4 × 3.25 × 9.36 mm after 10- and 30-second sonification, respectively. 30 s sonification with 118 W power at 50% duty cycle generated a peak temperature of 68 °C. Each ablation was visualized in real time by the built-in imaging probe.

Conclusion: We developed and validated an ultrasound-guided focused ultrasound probe for use in MIS procedures. The dimensional constraints of the prototype were designed to reflect those of BrainPath trocars, which are MIS tools used to create atraumatic access to deep-seated brain pathologies.
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http://dx.doi.org/10.1016/j.ultras.2020.106210DOI Listing
December 2020

Epidural Oscillating Cardiac-Gated Intracranial Implant Modulates Cerebral Blood Flow.

Neurosurgery 2020 Jun 13. Epub 2020 Jun 13.

Department of Mechanical Engineering, The University of Akron, Akron, Ohio.

Background: We have previously reported a method and device capable of manipulating ICP pulsatility while minimally effecting mean ICP.

Objective: To test the hypothesis that different modulations of the intracranial pressure (ICP) pulse waveform will have a differential effect on cerebral blood flow (CBF).

Methods: Using an epidural balloon catheter attached to a cardiac-gated oscillating pump, 13 canine subjects underwent ICP waveform manipulation comparing different sequences of oscillation in successive animals. The epidural balloon was implanted unilaterally superior to the Sylvian sulcus. Subjects underwent ICP pulse augmentation, reduction and inversion protocols, directly comparing time segments of system activation and deactivation. ICP and CBF were measured bilaterally along with systemic pressure and heart rate. CBF was measured using both thermal diffusion, and laser doppler probes.

Results: The activation of the cardiac-gate balloon implant resulted in an ipsilateral/contralateral ICP pulse amplitude increase with augmentation (217%/202% respectively, P < .0005) and inversion (139%/120%, P < .0005). The observed changes associated with the ICP mean values were smaller, increasing with augmentation (23%/31%, P < .0001) while decreasing with inversion (7%/11%, P = .006/.0003) and reduction (4%/5%, P < .0005). CBF increase was observed for both inversion and reduction protocols (28%/7.4%, P < .0001/P = .006 and 2.4%/1.3%, P < .0001/P = .003), but not the augmentation protocol. The change in CBF was correlated with ICP pulse amplitude and systolic peak changes and not with change in mean ICP or systemic variables (heart rate, arterial blood pressure).

Conclusion: Cardiac-gated manipulation of ICP pulsatility allows the study of intracranial pulsatile dynamics and provides a potential means of altering CBF.
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http://dx.doi.org/10.1093/neuros/nyaa188DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7666905PMC
June 2020

Evaluating the Effects of Cerebrospinal Fluid Protein Content on the Performance of Differential Pressure Valves and Antisiphon Devices Using a Novel Benchtop Shunting Model.

Neurosurgery 2020 10;87(5):1046-1054

Department of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland.

Background: Hydrocephalus is managed by surgically implanting flow-diversion technologies such as differential pressure valves and antisiphoning devices; however, such hardware is prone to failure. Extensive research has tested them in flow-controlled settings using saline or de-aerated water, yet little has been done to validate their performance in a setting recreating physiologically relevant parameters, including intracranial pressures, cerebrospinal fluid (CSF) protein content, and body position.

Objective: To more accurately chart the episodic drainage characteristics of flow-diversion technology. A gravity-driven benchtop model of flow was designed and tested continuously during weeks-long trials.

Methods: Using a hydrostatic pressure gradient as the sole driving force, interval flow rates of 6 valves were examined in parallel with various fluids. Daily trials in the upright and supine positions were run with fluid output collected from distal catheters placed at alternating heights for extended intervals.

Results: Significant variability in flow rates was observed, both within specific individual valves across different trials and among multiple valves of the same type. These intervalve and intravalve variabilities were greatest during supine trials and with increased protein. None of the valves showed evidence of overt obstruction during 30 d of exposure to CSF containing 5 g/L protein.

Conclusion: Day-to-day variability of ball-in-cone differential pressure shunt valves may increase overdrainage risk. Narrow-lumen high-resistance flow control devices as tested here under similar conditions appear to achieve more consistent flow rates, suggesting their use may be advantageous, and did not demonstrate any blockage or trend of decreasing flow over the 3 wk of chronic use.
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http://dx.doi.org/10.1093/neuros/nyaa203DOI Listing
October 2020

Evidence of Neural Microstructure Abnormalities in Type I Chiari Malformation: Associations Among Fiber Tract Integrity, Pain, and Cognitive Dysfunction.

Pain Med 2020 10;21(10):2323-2335

Department of Neurosurgery, Johns Hopkins Medical Center, Baltimore, Maryland, USA.

Background: Previous case-control investigations of type I Chiari malformation (CMI) have reported cognitive deficits and microstructural white matter abnormalities, as measured by diffusion tensor imaging (DTI). CMI is also typically associated with pain, including occipital headache, but the relationship between pain symptoms and microstructure is not known.

Methods: Eighteen CMI patients and 18 adult age- and education-matched control participants underwent DTI, were tested using digit symbol coding and digit span tasks, and completed a self-report measure of chronic pain. Tissue microstructure indices were used to examine microstructural abnormalities in CMI as compared with healthy controls. Group differences in DTI parameters were then reassessed after controlling for self-reported pain. Finally, DTI parameters were correlated with performance on the digit symbol coding and digit span tasks within each group.

Results: CMI patients exhibited greater fractional anisotropy (FA), lower radial diffusivity, and lower mean diffusivity in multiple brain regions compared with controls in diffuse white matter regions. Group differences no longer existed after controlling for self-reported pain. A significant correlation between FA and the Repeatable Battery for the Assessment of Neuropsychological Status coding performance was observed for controls but not for the CMI group.

Conclusions: Diffuse microstructural abnormalities appear to be a feature of CMI, manifesting predominantly as greater FA and less diffusivity on DTI sequences. These white matter changes are associated with the subjective pain experience of CMI patients and may reflect reactivity to neuroinflammatory responses. However, this hypothesis will require further deliberate testing in future studies.
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http://dx.doi.org/10.1093/pm/pnaa094DOI Listing
October 2020

Novel Risk Calculator for Suboccipital Decompression for Adult Chiari Malformation.

World Neurosurg 2020 07 29;139:526-534. Epub 2020 Apr 29.

Department of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA. Electronic address:

Background: Patient counseling and selection for surgical therapy in adult Chiari malformation type I (CM-1) remain debatable. We aimed to develop a clinical calculator predicting the risk of nonhome discharge and reoperation using the American College of Surgeons-National Surgical Quality Improvement Program database.

Methods: The database from years 2011 through 2017 was queried to identify the subset of CM-1 patients undergoing suboccipital decompression. Univariable analysis was conducted to identify baseline factors associated with nonhome discharge and 30-day reoperation following the initial decompression procedure. Logistic regression and the Akaike Information Criterion were used to identify the optimal models predictive of both outcomes. Performance was assessed using receiver operating curves and validated with bootstrapping.

Results: In 706 CM-1 patients, the rate of nonhome discharge was 5.2% and the reoperation rate was 6.6% with most reoperations consisting of cerebrospinal fluid flow diversion and cerebrospinal fluid leak repair. The optimal model predictive of nonhome discharge consisted of age (odds ratio [OR] = 1.05, P = 0.001), diabetes (OR = 2.44, P = 0.080), and American Society of Anesthesiologists class (OR = 1.94, P = 0.082) with an area under the curve of 0.720. The optimal model predictive of reoperation consisted of female sex (OR = 0.48, P = 0.031), body mass index (OR = 1.05, P = 0.002), and ASA class (OR = 3.44, P = 0.001) with an area under the curve of 0.726. A calculator for both outcomes was deployed under the following URL: https://jhuspine3.shinyapps.io/Discharge_Reop_Calculator/.

Conclusions: We have used a large international database to develop a simple risk calculator based on readily available preoperative variables. Following subsequent validation, this tool can help optimize patient counseling and decision making in adult CM-1.
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http://dx.doi.org/10.1016/j.wneu.2020.04.169DOI Listing
July 2020

Standardized regression-based clinical change score cutoffs for normal pressure hydrocephalus.

BMC Neurol 2020 Apr 16;20(1):140. Epub 2020 Apr 16.

Department of Neurology, Johns Hopkins University School of Medicine, 5200 Eastern Ave CTR STE 5100, Baltimore, MD, 21224, USA.

Background: Presently, for patients presenting with suspected Normal Pressure Hydrocephalus (NPH) who undergo temporary drainage of cerebrospinal fluid (CSF) there is no defined model to differentiate chance improvement form clinical significance change at the individual patient level. To address this lack of information we computed standard regression based clinical change models for the 10 Meter Walk Test, Timed Up & Go, Dual Timed Up & Go, 6-Minute Walk Test, Mini-Balance Evaluation Systems Test, Montreal Cognitive Assessment, and Symbol Digit Modalities using data from patients with suspected NPH that underwent temporary drainage of CSF. These clinically significant change modes can classify clinically significant improvement following temporary drainage of CSF at the individual patient level. This allows for physicians to differentiate a clinically significant improvement in symptoms from chance improvement.

Methods: Data was collected from 323 patients, over the age of 60, with suspected NPH that underwent temporary drainage of CSF with corresponding gait and cognitive testing. McSweeney Standardized Regression Based Clinical Change Models were computed for standard gait and cognitive measures: Timed Up & Go, Dual Timed Up & Go, 10 Meter Walk Test, MiniBESTest, 6-Minute Walk Test, Montreal Cognitive Assessment, and Symbol Digit Modalities Test. To assess the discriminate validity of the measures we used correlations, Chi, and regression analyses.

Results: The clinical change models explained 69-91.8% of the variability in post-drain performance (p <  0.001). As patient scores became more impaired, the percent change required for improvement to be clinically significant increased for all measures. We found that the measures were not discriminate, the Timed Up & Go was highly related to the 10 Meter Walk Test (r = 0.85, R = 0.769-0.738, p <  0.001), MiniBESTest (r = - 0.67, R = 0.589-0.734, p <  0.001), and 6 Minute Walk Test (r = - 0.77, R = 0.71-0.734, p <  0.001).

Conclusion: Standardized Regression Based Clinically Significant Change Models allow for physicians to use an evidence-based approach to differentiate clinically significant change from chance improvement at the individual patient level. The Timed Up & Go was shown to be predictive of detailed measures of gait velocity, balance, and endurance.
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http://dx.doi.org/10.1186/s12883-020-01719-yDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7164303PMC
April 2020

General Anesthesia Alters Intracranial Venous Pressures During Transverse Sinus Stenting.

World Neurosurg 2020 06 16;138:e712-e717. Epub 2020 Mar 16.

Department of Radiology, The Johns Hopkins Hospital, Baltimore, Maryland, USA. Electronic address:

Introduction: Pressure gradients across venous stenosis are used as a marker for physiologically significant narrowing in idiopathic intracranial hypertension. Performing such measurements under conscious sedation (CS) more likely reflects physiologic conditions, but can be uncomfortable, leading some operators to perform measurement under general anesthesia (GA), though this may not be equivalent.

Methods: We performed a retrospective analysis of patients who received endovascular transverse sinus stenting due to idiopathic intracranial hypertension between August 2013 and May 2017. Patients' demographics and anesthetic parameters were collected along with venous pressure measurements.

Results: We identified 15 patients (14 women). The mean (SD) age was 30.5 (9.0) years and the mean body mass index (SD) was 39.5 (9.6) kg/m. After measurements during CS, GA was induced with propofol and maintained with a volatile anesthetic. The median [IQR; range] transverse sinus pressure gradient under CS was 18 [12, 25; 6-38] mmHg compared with 14 [8, 21; 3-26] mm Hg under GA. The median [IQR; range] pressure gradient change after initiation of GA was -3 [-12, 0; -22 to 9] mm Hg (P = 0.014). After correction for increases in internal jugular vein pressures associated with assumption of GA, the median [IQR; range] gradient change was -11 [-12.5, -5; -22 to 0] mm Hg (P < 0.001).

Conclusions: The transition from CS to GA results in clinically meaningful reductions in transverse sinus gradients in idiopathic intracranial hypertension. Correction for increases in the internal jugular vein pressures reveals even more dramatic reductions in transverse sinus gradients.
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http://dx.doi.org/10.1016/j.wneu.2020.03.050DOI Listing
June 2020

First-In-Human Experience With Integration of Wireless Intracranial Pressure Monitoring Device Within a Customized Cranial Implant.

Oper Neurosurg (Hagerstown) 2020 09;19(3):341-350

Section of Neuroplastic and Reconstructive Surgery, Department of Plastic and Reconstructive Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland.

Background: Decompressive craniectomy is a lifesaving treatment for intractable intracranial hypertension. For patients who survive, a second surgery for cranial reconstruction (cranioplasty) is required. The effect of cranioplasty on intracranial pressure (ICP) is unknown.

Objective: To integrate the recently Food and Drug Administration-approved, fully implantable, noninvasive ICP sensor within a customized cranial implant (CCI) for postoperative monitoring in patients at high risk for intracranial hypertension.

Methods: A 16-yr-old female presented for cranioplasty 4-mo after decompressive hemicraniectomy for craniocerebral gunshot wound. Given the persistent transcranial herniation with concomitant subdural hygroma, there was concern for intracranial hypertension following cranioplasty. Thus, cranial reconstruction was performed utilizing a CCI with an integrated wireless ICP sensor, and noninvasive postoperative monitoring was performed.

Results: Intermittent ICP measurements were obtained twice daily using a wireless, handheld monitor. The ICP ranged from 2 to 10 mmHg in the supine position and from -5 to 4 mmHg in the sitting position. Interestingly, an average of 7 mmHg difference was consistently noted between the sitting and supine measurements.

Conclusion: This first-in-human experience demonstrates several notable findings, including (1) newfound safety and efficacy of integrating a wireless ICP sensor within a CCI for perioperative neuromonitoring; (2) proven restoration of normal ICP postcranioplasty despite severe preoperative transcranial herniation; and (3) proven restoration of postural ICP adaptations following cranioplasty. To the best of our knowledge, this is the first case demonstrating these intriguing findings with the potential to fundamentally alter the paradigm of cranial reconstruction.
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http://dx.doi.org/10.1093/ons/opz431DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7594174PMC
September 2020

Evidence for sex differences in morphological abnormalities in type I Chiari malformation.

Neuroradiol J 2019 Dec 18;32(6):458-466. Epub 2019 Jun 18.

Department of Biomedical Engineering, The University of Akron, USA.

Background And Purpose: Relatively little is known about the influence of individual difference variables on the presentation of macro-level brain morphology in type I Chiari malformation (CMI). The goal of the present study is to examine how case-control differences in Chiari are affected by patient sex.

Materials And Methods: Patient-provided magnetic resonance images were acquired through the Chiari 1000 database. Twenty-four morphometric measurements were taken using mid-sagittal images of 104 participants (26 male CMI, 26 female CMI, 26 male controls, and 26 female controls) using internally developed and validated custom software, Morphpro. Case-control comparisons were conducted separately by sex using healthy controls matched by age and body mass index. Probability-based -tests, effect sizes (Cohen's ), and confidence intervals were used to compare case-control differences separately by sex.

Results: Male and female case-control comparisons yielded largely the same trends of CMI-related morphometric abnormalities. Both groups yielded reductions in posterior cranial fossa (PCF) structure heights. However, there was evidence for greater PCF structure height reductions in male CMI patients as measured by Cohen's .

Conclusions: Case-control differences indicated strong consistency in the morphometric abnormalities of CMI malformation in males and females. However, despite the higher prevalence rates of CMI in females, the results from the present study suggest that male morphometric abnormalities may be greater in magnitude. These findings also provide insight into the inconsistent findings from previous morphometric studies of CMI and emphasize the importance of controlling for individual differences when conducting case-control comparisons in CMI.
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http://dx.doi.org/10.1177/1971400919857212DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6856994PMC
December 2019

The clinical spectrum of hydrocephalus in adults: report of the first 517 patients of the Adult Hydrocephalus Clinical Research Network registry.

J Neurosurg 2019 May 24;132(6):1773-1784. Epub 2019 May 24.

13Department of Clinical Neurosciences, Division of Neurosurgery, University of Calgary School of Medicine, Calgary, Alberta, Canada.

Objective: The authors describe the demographics and clinical characteristics of the first 517 patients enrolled in the Adult Hydrocephalus Clinical Research Network (AHCRN) during its first 2 years.

Methods: Adults ≥ 18 years were nonconsecutively enrolled in a registry at 6 centers. Four categories of adult hydrocephalus were defined: transition (treated before age 18 years), unrecognized congenital (congenital pattern, not treated before age 18 years), acquired (secondary to known risk factors, treated or untreated), and suspected idiopathic normal pressure hydrocephalus (iNPH) (≥ age 65 years, not previously treated). Data include etiology, symptoms, examination findings, neuropsychology screening, comorbidities, treatment, complications, and outcomes. Standard evaluations were administered to all patients by trained examiners, including the Montreal Cognitive Assessment, the Symbol Digit Modalities Test, the Beck Depression Inventory-II, the Overactive Bladder Questionnaire Short Form symptom bother, the 10-Meter Walk Test, the Boon iNPH gait scale, the Lawton Activities of Daily Living/Instrumental Activities of Daily Living (ADL/IADL) questionnaire, the iNPH grading scale, and the modified Rankin Scale.

Results: Overall, 517 individuals were enrolled. Age ranged from 18.1 to 90.7 years, with patients in the transition group (32.7 ± 10.0 years) being the youngest and those in the suspected iNPH group (76.5 ± 5.2 years) being the oldest. The proportion of patients in each group was as follows: 16.6% transition, 26.5% unrecognized congenital, 18.2% acquired, and 38.7% suspected iNPH. Excluding the 86 patients in the transition group, who all had received treatment, 79.4% of adults in the remaining 3 groups had not been treated at the time of enrollment. Patients in the suspected iNPH group had the poorest performance in cognitive evaluations, and those in the unrecognized congenital group had the best performance. The same pattern was seen in the Lawton ADL/IADL scores. Gait velocity was lowest in patients in the suspected iNPH group. Categories that had the most comorbidities (suspected iNPH) or etiologies of hydrocephalus that directly cause neurological injury (transition, acquired) had greater degrees of impairment compared to unrecognized congenital, which had the fewest comorbidities or etiologies associated with neurological injury.

Conclusions: The clinical spectrum of hydrocephalus in adults comprises more than iNPH or acquired hydrocephalus. Only 39% of patients had suspected iNPH, whereas 43% had childhood onset (i.e., those in the transition and unrecognized congenital groups). The severity of symptoms and impairment was worsened when the etiology of the hydrocephalus or complications of treatment caused additional neurological injury or when multiple comorbidities were present. However, more than half of patients in the transition, unrecognized congenital, and acquired hydrocephalus groups had minimal or no impairment. Excluding the transition group, nearly 80% of patients in the AHCRN registry were untreated at the time of enrollment. A future goal for the AHCRN is to determine whether patients with unrecognized congenital and acquired hydrocephalus need treatment and which patients in the suspected iNPH cohort actually have possible hydrocephalus and should undergo further diagnostic testing. Future prospective research is needed in the diagnosis, treatment, outcomes, quality of life, and macroeconomics of all categories of adult hydrocephalus.
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http://dx.doi.org/10.3171/2019.2.JNS183538DOI Listing
May 2019

Type I Chiari malformation, RBANS performance, and brain morphology: Connecting the dots on cognition and macrolevel brain structure.

Neuropsychology 2019 Jul 16;33(5):725-738. Epub 2019 May 16.

Department of Neurosurgery.

Background: Idiopathic descent of cerebellar tonsils into the cervical spine in Chiari malformation Type I (CMI) is typically associated with occipital headache. Accumulating evidence from experimental studies suggests cognitive effects of CMI. The aim of the current study was to examine the relationship between cognition and CMI using a battery of standardized neuropsychological and symptom inventory instruments.

Method: Eighteen untreated adults with CMI, and 18 gender, age, and education matched healthy controls completed the Repeatable Battery for the Assessment of Neuropsychological Status (RBANS), and standardized measures of pain, mood, and disability. Morphometric measurements of key neural and osseous elements were also obtained from structural brain magnetic resonance images, for correlation with symptom outcomes.

Results: CMI patients exhibited deficits in RBANS attention, immediate memory, delayed memory, and total score. After controlling for pain and associated affective disturbance, the significant group effect for RBANS attention remained. CMI patients also presented seven morphometric differences comprising the cerebellum and posterior cranial fossa compartment that differed from healthy controls, some of which were associated with self-reported pain and disability. Notably, group differences in tonsillar position were associated with self-reported pain, disability, and delayed memory.

Conclusion: Adult CMI is associated with domain-specific cognitive change, detectable using a standard clinical instrument. The extent of cognitive impairment is independent of pain or affective symptomatology and may be related to the key pathognomonic feature of the condition. (PsycINFO Database Record (c) 2019 APA, all rights reserved).
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http://dx.doi.org/10.1037/neu0000547DOI Listing
July 2019

First-in-Human Experience With Integration of a Hydrocephalus Shunt Device Within a Customized Cranial Implant.

Oper Neurosurg (Hagerstown) 2019 12;17(6):608-615

Department of Neurosurgery, Johns Hopkins University School of Medicine, The Johns Hopkins Hospital, Baltimore, Maryland.

Background: Implantable shunt devices are critical and life saving for hydrocephalus patients. However, these devices are fraught with high complication rates including scalp dehiscence, exposure, and extrusion. In fact, high shunt valve profiles are correlated with increased complications compared to those with lower profiles. As such, we sought a new method for integrating shunt valves for those challenging patients presenting with scalp-related complications.

Objective: To safely implant and integrate a hydrocephalus shunt valve device within a customized cranial implant, in an effort to limit its high-profile nature as a main contributor to shunt failure and scalp breakdown, and at the same time, improve patient satisfaction by preventing contour deformity.

Methods: A 64-yr-old male presented with an extruding hydrocephalus shunt valve and chronic, open scalp wound. The shunt valve was removed and temporary shunt externalization was performed. He received 2 wk of culture-directed antibiotics. Next, a contralateral craniectomy was performed allowing a new shunt valve system to be implanted within a low-profile, customized cranial implant. All efforts were made, at the patient's request, to decrease the high-profile nature of the shunt valve contributing to his most recent complication.

Results: First-in-human implantation was performed without complication. Postoperative shunt identification and programming was uncomplicated. The high-profile nature of the shunt valve was decreased by 87%. At 10 mo, the patient has experienced no complications and is extremely satisfied with his appearance.

Conclusion: This first-in-human experience suggests that a high-profile hydrocephalus shunt device may be safely integrated within a customized cranial implant.
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http://dx.doi.org/10.1093/ons/opz003DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6855953PMC
December 2019

Development of Common Data Elements for Use in Chiari Malformation Type I Clinical Research: An NIH/NINDS Project.

Neurosurgery 2019 12;85(6):854-860

Division of Neuroscience, National Institutes of Health/National Institute of Neurological Disorders and Stroke, Bethesda, Maryland.

The management of Chiari I malformation (CMI) is controversial because treatment methods vary and treatment decisions rest on incomplete understanding of its complex symptom patterns, etiologies, and natural history. Validity of studies that attempt to compare treatment of CMI has been limited because of variable terminology and methods used to describe study subjects. The goal of this project was to standardize terminology and methods by developing a comprehensive set of Common Data Elements (CDEs), data definitions, case report forms (CRFs), and outcome measure recommendations for use in CMI clinical research, as part of the CDE project at the National Institute of Neurological Disorders and Stroke (NINDS) of the US National Institutes of Health. A working group, comprising over 30 experts, developed and identified CDEs, template CRFs, data dictionaries, and guidelines to aid investigators starting and conducting CMI clinical research studies. The recommendations were compiled, internally reviewed, and posted online for external public comment. In October 2016, version 1.0 of the CMI CDE recommendations became available on the NINDS CDE website. The recommendations span these domains: Core Demographics/Epidemiology; Presentation/Symptoms; Co-Morbidities/Genetics; Imaging; Treatment; and Outcome. Widespread use of CDEs could facilitate CMI clinical research trial design, data sharing, retrospective analyses, and consistent data sharing between CMI investigators around the world. Updating of CDEs will be necessary to keep them relevant and applicable to evolving research goals for understanding CMI and its treatment.
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http://dx.doi.org/10.1093/neuros/nyy475DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7054710PMC
December 2019

Cerebellar tonsil ectopia measurement in type I Chiari malformation patients show poor inter-operator reliability.

Fluids Barriers CNS 2018 Dec 17;15(1):33. Epub 2018 Dec 17.

Department of Biological Engineering, University of Idaho, 875 Perimeter Drive MS 0904, Moscow, ID, 83844-0904, USA.

Background: Type 1 Chiari malformation (CM-I) has been historically defined by cerebellar tonsillar position (TP) greater than 3-5 mm below the foramen magnum (FM). Often, the radiographic findings are highly variable, which may influence the clinical course and patient outcome. In this study, we evaluate the inter-operator reliability (reproducibility) of MRI-based measurement of TP in CM-I patients and healthy controls.

Methods: Thirty-three T2-weighted MRI sets were obtained for 23 CM-I patients (11 symptomatic and 12 asymptomatic) and 10 healthy controls. TP inferior to the FM was measured in the mid-sagittal plane by seven expert operators with reference to McRae's line. Overall agreement between the operators was quantified by intraclass correlation coefficient (ICC).

Results: The mean and standard deviation of cerebellar TP measurements for asymptomatic (CM-Ia) and symptomatic (CM-Is) patients in mid-sagittal plane was 6.38 ± 2.19 and 9.57 ± 2.63 mm, respectively. TP measurements for healthy controls was 0.48 ± 2.88 mm. The average range of TP measurements for all data sets analyzed was 7.7 mm. Overall operator agreement for TP measurements was relatively high with an ICC of 0.83.

Conclusion: The results demonstrated a large average range (7.7 mm) of measurements among the seven expert operators and support that, if economically feasible, two radiologists should make independent measurements before radiologic diagnosis of CM-I and surgery is contemplated. In the future, an objective diagnostic parameter for CM-I that utilizes automated algorithms and results in smaller inter-operator variation may improve patient selection.
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http://dx.doi.org/10.1186/s12987-018-0118-1DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6296028PMC
December 2018

Evaluation of an in vivo model for ventricular shunt infection: a pilot study using a novel antimicrobial-loaded polymer.

J Neurosurg 2018 08;131(2):587-595

Departments of1Neurosurgery.

Objective: Ventricular shunt infection remains an issue leading to high patient morbidity and cost, warranting further investigation. The authors sought to create an animal model of shunt infection that could be used to evaluate possible catheter modifications and innovations.

Methods: Three dogs underwent bilateral ventricular catheter implantation and inoculation with methicillin-sensitive Staphylococcus aureus (S. aureus). In 2 experimental animals, the catheters were modified with a polymer containing chemical "pockets" loaded with vancomycin. In 1 control animal, the catheters were polymer coated but without antibiotics. Animals were monitored for 9 to 11 days, after which the shunts were explanted. MRI was performed after shunt implantation and prior to catheter harvest. The catheters were sonicated prior to microbiological culture and also evaluated by electron microscopy. The animals' brains were evaluated for histopathology.

Results: All animals underwent successful catheter implantation. The animals developed superficial wound infections, but no neurological deficits. Imaging demonstrated ventriculitis and cerebral edema. Harvested catheters from the control animal demonstrated > 104 colony-forming units (CFUs) of S. aureus. In the first experimental animal, one shunt demonstrated > 104 CFUs of S. aureus, but the other demonstrated no growth. In the second experimental animal, one catheter demonstrated no growth, and the other grew trace S. aureus. Brain histopathology revealed acute inflammation and ventriculitis in all animals, which was more severe in the control.

Conclusions: The authors evaluated an animal model of ventricular shunting and reliably induced features of shunt infection that could be microbiologically quantified. With this model, investigation of pathophysiological and imaging correlates of infection and potentially beneficial shunt catheter modifications is possible.
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http://dx.doi.org/10.3171/2018.1.JNS172523DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6677638PMC
August 2018

Cardiac-Related Spinal Cord Tissue Motion at the Foramen Magnum is Increased in Patients with Type I Chiari Malformation and Decreases Postdecompression Surgery.

World Neurosurg 2018 Aug 4;116:e298-e307. Epub 2018 May 4.

Department of Biological Engineering, University of Idaho, Moscow, Idaho, USA. Electronic address:

Objective: Type 1 Chiari malformation (CM-I) is a craniospinal disorder historically defined by cerebellar tonsillar position greater than 3-5 mm below the foramen magnum (FM). This definition has come under question because quantitative measurements of cerebellar herniation do not always correspond with symptom severity. Researchers have proposed several additional radiographic diagnostic criteria based on dynamic motion of fluids and/or tissues. The present study objective was to determine if cardiac-related craniocaudal spinal cord tissue displacement is an accurate indicator of the presence of CM-I and if tissue displacement is altered with decompression.

Methods: A cohort of 20 symptomatic patients underwent decompression surgery. Fifteen healthy volunteers were recruited for comparison with the CM-I group. Axial phase-contrast magnetic resonance imaging (PC-MRI) measurements were collected before and after surgery at the FM with cranial-caudal velocity encoding and 20 frames per cardiac cycle with retrospective reconstruction. Spinal cord motion (SCM) at the FM was quantified based on the peak-to-peak integral of average spinal cord velocity.

Results: Tissue motion for the presurgical group was significantly greater than controls (P = 0.0009). Motion decreased after surgery (P = 0.058) with an effect size of -0.151 mm and a standard error of 0.066 mm. Postoperatively, no statistical difference from controls in bulk displacement at the FM was found (P = 0.200) after post hoc testing using the Tukey adjustment for multiple comparisons.

Conclusions: These results support SCM measurement by PC-MRI as a possible noninvasive radiographic diagnostic for CM-I. Dynamic measurement of SCM provides unique diagnostic information about CM-I alongside static quantification of tonsillar position and other intracranial morphometrics.
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http://dx.doi.org/10.1016/j.wneu.2018.04.191DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6063776PMC
August 2018
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