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Kevin K. Kumar, Linden E. Fornoff, Robert L. Dodd, Michael P. Marks, and David S. Hong

BACKGROUND

Vein of Galen aneurysmal malformations (VGAMs) are rare congenital intracranial vascular lesions that represent 30% of all pediatric vascular anomalies. These lesions are associated with severe manifestations, including congestive heart failure, hydrocephalus, and spontaneous hemorrhage. The mainstay of management is medical stabilization followed by endovascular embolization of the lesion. Although VGAM was first reported in 1937, there are few published cases demonstrating spontaneous regression of the lesion.

OBSERVATIONS

The authors report the case of a 31-month-old female who presented with an incidentally found VGAM. After initial evaluation, including magnetic resonance imaging and angiography, the patient was lost to follow-up. Upon her return to the clinic at age 12 years, the previously identified VGAM was absent, indicative of involution of the lesion. The patient remained asymptomatic and met appropriate developmental milestones during this interval.

LESSONS

This report adds a rare case of the spontaneous resolution of VGAM to the literature. This case may suggest the presence of VGAMs that are asymptomatic, undetected, and regress within the pediatric population. Future studies may benefit from identifying imaging and angiographic findings predictive of spontaneous regression. There may be a role for conservative management in particular cases of asymptomatic and medically stable children with VGAMs.

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Michael K.-Y. Hong, Matthew K.-H. Hong, Wei-Ren Pan, David Wallace, Mark W. Ashton, and G. Ian Taylor

Object

The angiosome concept has been the subject of extensive research by the senior author (G.I.T.), but its specific applicability to the spinal cord was hitherto unknown. The aim of this study was to see if the spinal cord vasculature followed the angiosome concept and to review the usefulness of preoperative spinal angiography in surgery for spinal disorders. Spinal cord infarction and permanent paraplegia may result from inadvertent interruption of the artery of Adamkiewicz. Spinal angiography, which may enable avoidance of this catastrophic complication, is still not commonly used.

Methods

Two fresh cadavers were injected with a gelatin–lead oxide mixture for detailed comparative study of spinal cord vasculature. One cadaver had insignificant vascular disease, whereas the other had extensive aortic atherosclerosis, presenting a unique opportunity for study. After removal from each cadaver, radiographs of the spinal cords were obtained, then photographed, and the vascular territories of the cords were defined.

Results

Four angiosome territories were defined: vertebral, subclavian, posterior intercostal, and lumbar. These vascular territories were joined longitudinally by true anastomotic channels along the anterior and posterior spinal cord. Anastomosis between the anterior and posterior vasculature was poor in the thoracolumbar region. The anterior cord relied on fewer feeder arteries than the posterior, and the anterior thoracolumbar cord depended on the artery of Adamkiewicz for its supply. In chronic aortic disease with intercostal artery occlusion at multiple levels, a rich collateral circulation supporting the spinal cord was found.

Conclusions

The arterial supply of the spinal cord follows the angiosome concept. The atherosclerotic specimen supports the suggestion that the blood supply is able to adapt to gradual vascular occlusion through development of a collateral circulation. Nevertheless, the spinal cord is susceptible to ischemia when faced with acute vascular occlusion. This includes inadvertent interruption of the artery of Adamkiewicz. The authors recommend the use of preoperative spinal angiography to prevent possible paraplegia in removal of thoracolumbar spinal tumors.

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Matthias Oertel, Daniel F. Kelly, David McArthur, W. John Boscardin, Thomas C. Glenn, Jae Hong Lee, Tooraj Gravori, Dennis Obukhov, Duncan Q. McBride, and Neil A. Martin

Object. Progressive intracranial hemorrhage after head injury is often observed on serial computerized tomography (CT) scans but its significance is uncertain. In this study, patients in whom two CT scans were obtained within 24 hours of injury were analyzed to determine the incidence, risk factors, and clinical significance of progressive hemorrhagic injury (PHI).

Methods. The diagnosis of PHI was determined by comparing the first and second CT scans and was categorized as epidural hematoma (EDH), subdural hematoma (SDH), intraparenchymal contusion or hematoma (IPCH), or subarachnoid hemorrhage (SAH). Potential risk factors, the daily mean intracranial pressure (ICP), and cerebral perfusion pressure were analyzed. In a cohort of 142 patients (mean age 34 ± 14 years; median Glasgow Coma Scale score of 8, range 3–15; male/female ratio 4.3:1), the mean time from injury to first CT scan was 2 ± 1.6 hours and between first and second CT scans was 6.9 ± 3.6 hours. A PHI was found in 42.3% of patients overall and in 48.6% of patients who underwent scanning within 2 hours of injury. Of the 60 patients with PHI, 87% underwent their first CT scan within 2 hours of injury and in only one with PHI was the first CT scan obtained more than 6 hours postinjury. The likelihood of PHI for a given lesion was 51% for IPCH, 22% for EDH, 17% for SAH, and 11% for SDH. Of the 46 patients who underwent craniotomy for hematoma evacuation, 24% did so after the second CT scan because of findings of PHI. Logistic regression was used to identify male sex (p = 0.01), older age (p = 0.01), time from injury to first CT scan (p = 0.02), and initial partial thromboplastin time (PTT) (p = 0.02) as the best predictors of PHI. The percentage of patients with mean daily ICP greater than 20 mm Hg was higher in those with PHI compared with those without PHI. The 6-month postinjury outcome was similar in the two patient groups.

Conclusions. Early progressive hemorrhage occurs in almost 50% of head-injured patients who undergo CT scanning within 2 hours of injury, it occurs most frequently in cerebral contusions, and it is associated with ICP elevations. Male sex, older age, time from injury to first CT scan, and PTT appear to be key determinants of PHI. Early repeated CT scanning is indicated in patients with nonsurgically treated hemorrhage revealed on the first CT scan.

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Derek Yecies, Katie Shpanskaya, Rashad Jabarkheel, Maryam Maleki, Lisa Bruckert, Samuel H. Cheshier, David Hong, Michael S. B. Edwards, Gerald A. Grant, and Kristen W. Yeom

OBJECTIVE

Posterior fossa syndrome (PFS) is a common complication following the resection of posterior fossa tumors in children. The pathophysiology of PFS remains incompletely elucidated; however, the wide-ranging symptoms of PFS suggest the possibility of widespread cortical dysfunction. In this study, the authors utilized arterial spin labeling (ASL), an MR perfusion modality that provides quantitative measurements of cerebral blood flow without the use of intravenous contrast, to assess cortical blood flow in patients with PFS.

METHODS

A database of medulloblastoma treated at the authors’ institution from 2004 to 2016 was retrospectively reviewed, and 14 patients with PFS were identified. Immediate postoperative ASL for patients with PFS and medulloblastoma patients who did not develop PFS were compared. Additionally, in patients with PFS, ASL following the return of speech was compared with immediate postoperative ASL.

RESULTS

On immediate postoperative ASL, patients who subsequently developed PFS had statistically significant decreases in right frontal lobe perfusion and a trend toward decreased perfusion in the left frontal lobe compared with controls. Patients with PFS had statistically significant increases in bilateral frontal lobe perfusion after the resolution of symptoms compared with their immediate postoperative imaging findings.

CONCLUSIONS

ASL perfusion imaging identifies decreased frontal lobe blood flow as a strong physiological correlate of PFS that is consistent with the symptomatology of PFS. This is the first study to demonstrate that decreases in frontal lobe perfusion are present in the immediate postoperative period and resolve with the resolution of symptoms, suggesting a physiological explanation for the transient symptoms of PFS.

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Solomiia Savchuk, Michael C. Jin, Stephanie Choi, Lily H. Kim, Jennifer L. Quon, Anthony Bet, Laura M. Prolo, David S. Hong, Kelly B. Mahaney, and Gerald A. Grant

OBJECTIVE

Optimal management of pediatric Chiari malformation type I (CM-I) is much debated, chiefly due to the lack of validated tools for outcome assessment, with very few tools incorporating patient-centered measures of health-related quality of life (HRQOL). Although posterior fossa decompression (PFD) benefits a subset of patients, prediction of its impact across patients is challenging. The primary aim of this study was to investigate the role of patient-centered HRQOL measures in the assessment and prediction of outcomes after PFD.

METHODS

The authors collected HRQOL data from a cohort of 20 pediatric CM-I patients before and after PFD. The surveys included assessments of selected Patient-Reported Outcomes Measurement Information System (PROMIS) health domains and were used to generate the PROMIS preference (PROPr) score, which is a measure of HRQOL. PROMIS is a reliable standardized measure of HRQOL domains such as pain, fatigue, depression, and physical function, which are all relevant to CM-I. The authors then compared the PROPr scores with Chicago Chiari Outcome Scale (CCOS) scores derived from time-matched clinical documentation. Finally, the authors used the PROPr scores as an outcome measure to predict postsurgical HRQOL improvement at 1 year on the basis of patient demographic characteristics, comorbidities, and radiological and physical findings. The Wilcoxon signed-rank test, Mann-Whitney U-test, and Kendall’s correlation were used for statistical analysis.

RESULTS

Aggregate analysis revealed improvement of pain severity after PFD (p = 0.007) in anatomical patterns characteristic of CM-I. Most PROMIS domain scores trended toward improvement after surgery, with anxiety and pain interference reaching statistical significance (p < 0.002 and p < 0.03, respectively). PROPr scores also significantly improved after PFD (p < 0.008). Of the baseline patient characteristics, preexisting scoliosis was the most accurate negative predictor of HRQOL improvement after PFD (median −0.095 vs 0.106, p < 0.001). A correlation with modest magnitude (Kendall’s tau range 0.19–0.47) was detected between the patient-centered measures and CCOS score.

CONCLUSIONS

The authors observed moderate improvement of HRQOL, when measured using a modified panel of PROMIS question banks, in this pilot cohort of pediatric CM-I patients after PFD. Further investigations are necessary to validate this tool for children with CM-I and to determine whether these scores correlate with clinical and radiographic findings.

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Thu-Trang Hickman, Matthew E. Shuman, Tatyana A. Johnson, Felix Yang, Rebecca R. Rice, Isaac M. Rice, Esther H. Chung, Robert Wiemann, Megan Tinl, Christine Iracheta, Grace Chen, Patricia Flynn, Mary Beth Mondello, Jillian Thompson, Mary-Ellen Meadows, Rona S. Carroll, Hong Wei Yang, Hongyan Xing, David Pilgrim, E. Antonio Chiocca, Ian F. Dunn, Alexandra J. Golby, and Mark D. Johnson

OBJECTIVE

Idiopathic normal pressure hydrocephalus (iNPH) is characterized by ventriculomegaly, gait difficulty, incontinence, and dementia. The symptoms can be ameliorated by CSF drainage. The object of this study was to identify factors associated with shunt-responsive iNPH.

METHODS

The authors reviewed the medical records of 529 patients who underwent shunt placement for iNPH at their institution between July 2001 and March 2015. Variables associated with shunt-responsive iNPH were identified using bivariate and multivariate analyses. Detailed alcohol consumption information was obtained for 328 patients and was used to examine the relationship between alcohol and shunt-responsive iNPH. A computerized patient registry from 2 academic medical centers was queried to determine the prevalence of alcohol abuse among 1665 iNPH patients.

RESULTS

Bivariate analysis identified associations between shunt-responsive iNPH and gait difficulty (OR 4.59, 95% CI 2.32–9.09; p < 0.0001), dementia (OR 1.79, 95% CI 1.14–2.80; p = 0.01), incontinence (OR 1.77, 95% CI 1.13–2.76; p = 0.01), and alcohol use (OR 1.98, 95% CI 1.23–3.16; p = 0.03). Borderline significance was observed for hyperlipidemia (OR 1.56, 95% CI 0.99–2.45; p = 0.054), a family history of hyperlipidemia (OR 3.09, 95% CI 0.93–10.26, p = 0.054), and diabetes (OR 1.83, 95% CI 0.96–3.51; p = 0.064). Multivariate analysis identified associations with gait difficulty (OR 3.98, 95% CI 1.81–8.77; p = 0.0006) and alcohol (OR 1.94, 95% CI 1.10–3.39; p = 0.04). Increased alcohol intake correlated with greater improvement after CSF drainage. Alcohol abuse was 2.5 times more prevalent among iNPH patients than matched controls.

CONCLUSIONS

Alcohol consumption is associated with the development of shunt-responsive iNPH.