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1.
J Neurotrauma ; 40(13-14): 1451-1458, 2023 07.
Article in English | MEDLINE | ID: mdl-36517974

ABSTRACT

Blunt cerebrovascular injury (BCVI) is defined as blunt trauma to the head and neck leading to damage to the vertebral and/or carotid arteries; debate exists regarding which children are considered at high risk for BCVI and in need of angiographic/vessel imaging. We previously proposed a screening tool, the McGovern score, to identify pediatric trauma patients at high risk for BCVI, and we aim to validate the McGovern score by pooling data from multiple pediatric trauma centers. This is a multi-center, hospital-based, cohort study from all prospectively registered pediatric (<16 years of age) trauma patients who presented to the emergency department (ED) between 2003 and 2017 at six Level 1 pediatric trauma centers. The registry was retrospectively queried for patients who received a computed tomography angiogram (CTA) as a screening method for BCVI. Age, length of follow-up, mechanism of injury (MOI), arrival Glasgow Coma Scale (GCS) score, and focal neurological deficit were recorded. Radiological variables queried were the presence of a carotid canal fracture, petrous temporal bone fracture, and CT presence of infarction. Patients with BCVI were queried for mode of treatment, type of intracranial injury, artery damaged, and BCVI injury grade. The McGovern score was calculated for all patients who underwent CTA across all data groups. A total of 1012 patients underwent CTA; 72 of these patients were found to have BCVI, 51 of which were in the validation cohort. Across all data groups, the McGovern score has a >80% sensitivity (SN) and >98% negative predictive value (NPV). The McGovern score for pediatric BCVI is an effective, generalizable screening tool.


Subject(s)
Cerebrovascular Trauma , Multiple Trauma , Wounds, Nonpenetrating , Humans , Child , Cohort Studies , Retrospective Studies , Cerebrovascular Trauma/diagnostic imaging , Wounds, Nonpenetrating/diagnostic imaging , Tomography, X-Ray Computed
2.
J Neurosurg Pediatr ; : 1-8, 2022 May 13.
Article in English | MEDLINE | ID: mdl-35561696

ABSTRACT

OBJECTIVE: Caregiver stress from a child's diagnosis can impact a caregiver's ability to participate in treatment decisions, comply, and manage long-term illness. The aim of this study was to compare caregiver stress in children with craniosynostosis at diagnosis and postoperatively. METHODS: This prospective study included caregivers of pediatric patients with craniosynostosis receiving operative intervention. Demographics and Parenting Stress Index, Short Form (PSI-SF) and Pediatric Inventory for Parents (PIP) surveys at baseline (preoperatively) and 3 and 6 months postoperatively were completed. PSI-SF scores between 15 and 80 are considered normal, with > 85 being clinically significant and requiring follow-up. Higher PIP scores represent increased frequency and difficulty of stressful events due to the child's illness. Pairwise comparisons were performed using the Wilcoxon signed-rank test. Multivariate analysis was performed to assess for PSI-SF and PIP predictors. RESULTS: Of 106 caregivers (84% Caucasian), there were 62 mothers and 40 fathers. There were 68 and 45 responses at 3 and 6 months postoperatively, respectively. Regarding the baseline group, more than 80% were between 20 and 40 years of age and 58% had less than 2 years of college education. The median household income fell in the $45,001-$60,000 bracket. There was no significant difference between median baseline PSI-SF score (65, IQR 51-80) and those at 3 months (p = 0.45) and 6 months (p = 0.82) postoperatively. Both median PIP frequency (89 vs 74, p < 0.01) and difficulty (79 vs 71, p < 0.01) scores were lower at 3 months, although no significant difference was observed at 6 months (frequency: 95 vs 91, p = 0.67; difficulty: 82 vs 80, p = 0.34). Female sex, uninsured status, and open surgery type were all risk factors for higher parental stress. CONCLUSIONS: Stress levels ranged from normal to clinically significant in the caregivers, with sex, uninsured status, and open repair predicting higher stress. Stress decreased at 3 months postoperatively before increasing at 6 months. Intervention targeting caregiver stress should be explored to maintain lower stress observed at 3 months after surgery.

3.
Phys Sportsmed ; 50(2): 171-180, 2022 04.
Article in English | MEDLINE | ID: mdl-33764271

ABSTRACT

OBJECTIVE: Sport participation promotes health benefits for children. Current media and scientific coverage of sport-related head injury may influence a parent's decision on sports participation. Physicians must understand what influences these decisions to effectively counsel patients and families. This study sought to better understand and quantify the parental decisions to allow/disallow collision sports, including football participation. METHODS: A 31-question survey related to child/parent demographics, sports history, and influences to allow/disallow sport participation was available to parents in the United States through a national volunteer registry, between November 2016 and September 2019. Pearson's chi-squared and Wilcoxon's signed-rank test were used to analyze categorical and continuous variables, respectively. Multivariate logistic regression was performed to identify the most powerful factors associated with the decision. RESULTS: Of the 884 responses, 430 (49%) parents would disallow collision sport participation and 334 (38%) would disallow football. Parents who would allow collision sports more commonly cited child desire, while those parents who would disallow cited safety concern as the greatest influence to generally disallow a sport. Those who would disallow reported doctors (35.1% vs 25.3%; P = 0.002), media (12.8% vs 7.3%; P = 0.006) and other parents (11.2% vs 7.3%; P = 0.045) as influences. A child's age (OR 1.039, 95%CI 1.007-1.073; P = 0.018) was independently associated with their parent responding that they would allow collision sports. Parent educational status showed that those with higher than a bachelor's degree would be less likely to allow football participation (OR 0.635, 95%CI 0.443-0.910; P = 0.013). CONCLUSIONS: Parents incorporate many sources of information into the decision to allow or disallow their child to participate in collision sports. A child's desire to play a sport and child safety are driving factors for parents faced with the decision of whether to allow participation in collision sports. Physician input is more frequently influential to parents than the media, underscoring the responsibility of physicians to engage families on the risks, benefits, and resources available for sport participation.


Subject(s)
Athletic Injuries , Football , Soccer , Athletic Injuries/epidemiology , Athletic Injuries/prevention & control , Child , Humans , Logistic Models , Parents , Surveys and Questionnaires , United States/epidemiology
7.
Am J Hum Genet ; 108(1): 100-114, 2021 01 07.
Article in English | MEDLINE | ID: mdl-33352116

ABSTRACT

Chiari I malformation (CM1), the displacement of the cerebellum through the foramen magnum into the spinal canal, is one of the most common pediatric neurological conditions. Individuals with CM1 can present with neurological symptoms, including severe headaches and sensory or motor deficits, often as a consequence of brainstem compression or syringomyelia (SM). We conducted whole-exome sequencing (WES) on 668 CM1 probands and 232 family members and performed gene-burden and de novo enrichment analyses. A significant enrichment of rare and de novo non-synonymous variants in chromodomain (CHD) genes was observed among individuals with CM1 (combined p = 2.4 × 10-10), including 3 de novo loss-of-function variants in CHD8 (LOF enrichment p = 1.9 × 10-10) and a significant burden of rare transmitted variants in CHD3 (p = 1.8 × 10-6). Overall, individuals with CM1 were found to have significantly increased head circumference (p = 2.6 × 10-9), with many harboring CHD rare variants having macrocephaly. Finally, haploinsufficiency for chd8 in zebrafish led to macrocephaly and posterior hindbrain displacement reminiscent of CM1. These results implicate chromodomain genes and excessive brain growth in CM1 pathogenesis.


Subject(s)
Arnold-Chiari Malformation/genetics , DNA-Binding Proteins/genetics , Polymorphism, Single Nucleotide/genetics , Adult , Animals , Arnold-Chiari Malformation/pathology , Brain/pathology , Case-Control Studies , Female , Haploinsufficiency/genetics , Humans , Magnetic Resonance Imaging/methods , Male , Syringomyelia/genetics , Exome Sequencing/methods , Zebrafish/genetics
8.
Childs Nerv Syst ; 37(1): 47-54, 2021 01.
Article in English | MEDLINE | ID: mdl-32468243

ABSTRACT

PURPOSE: Blunt cerebrovascular injury (BCVI) is uncommon in the pediatric population. Among the management options is medical management consisting of antithrombotic therapy with either antiplatelets or anticoagulation. There is no consensus on whether administration of antiplatelets or anticoagulation is more appropriate for BCVI in children < 10 years of age. Our goal was to compare radiographic and clinical outcomes based on medical treatment modality for BCVI in children < 10 years. METHODS: Clinical and radiographic data were collected retrospectively for children screened for BCVI with computed tomography angiography at 5 academic pediatric trauma centers. RESULTS: Among 651 patients evaluated with computed tomography angiography to screen for BCVI, 17 patients aged less than 10 years were diagnosed with BCVI (7 grade I, 5 grade II, 1 grade III, 4 grade IV) and received anticoagulation or antiplatelet therapy for 18 total injuries: 11 intracranial carotid artery, 4 extracranial carotid artery, and 3 extracranial vertebral artery injuries. Eleven patients were treated with antiplatelets (10 aspirin, 1 clopidogrel) and 6 with anticoagulation (4 unfractionated heparin, 2 low-molecular-weight heparin, 1 transitioned from the former to the latter). There were no complications secondary to treatment. One patient who received anticoagulation died as a result of the traumatic injuries. In aggregate, children treated with antiplatelet therapy demonstrated healing on 52% of follow-up imaging studies versus 25% in the anticoagulation cohort. CONCLUSION: There were no observed differences in the rate of hemorrhagic complications between anticoagulation and antiplatelet therapy for BCVI in children < 10 years, with a nonsignificantly better rate of healing on follow-up imaging in children who underwent antiplatelet therapy; however, the study cohort was small despite including patients from 5 hospitals.


Subject(s)
Platelet Aggregation Inhibitors , Wounds, Nonpenetrating , Anticoagulants/therapeutic use , Child , Cohort Studies , Heparin , Humans , Platelet Aggregation Inhibitors/therapeutic use , Retrospective Studies
9.
J Neurointerv Surg ; 13(7): 661-668, 2021 Jul.
Article in English | MEDLINE | ID: mdl-33077576

ABSTRACT

BACKGROUND: Identifying drivers of nationwide variation in healthcare costs could help reduce overall cost. Endovascular treatment for unruptured cerebral aneurysms (ETUCR) is an elective neurointerventional procedure that allows for detailed analysis of cost variation. This study aimed to investigate the role of insurance type in cost variation of ETUCR. METHODS: A retrospective analysis of patients undergoing ETUCR was done. Demographic and hospital data were obtained from the National Inpatient Sample 2012-2015. Multivariate analysis was done using a generalized linear model. Oaxaca-Blinder decomposition was performed to identify factors driving cost variation. RESULTS: There was a significant difference in median cost ($25 331.82 vs $25 825.25, respectively, P<0.001) as well as length of stay (P<0.001) and complications (P<0.001) between patients with private insurance and Medicare. In multivariate analysis, insurance type was not predictive of increased cost. Among patients aged 65-75 years there was a higher median cost with private insurance compared to Medicare ($28 373.85 vs $25 558.25, respectively, P<0.001) but no difference in complications or length of stay. Oaxaca-Blinder decomposition showed higher marginal costs associated with private insurance patients at hospitals with greater endovascular operative volume (P=0.015). CONCLUSIONS: In patients aged 65-75 years, private insurance is associated with higher costs compared to Medicare; however, insurance type is not predictive of increased cost in multivariate analysis. Differential treatment of private insurance and Medicare patients at hospitals with greater operative volume seems to influence this difference, likely due to differential reimbursement schemes that lead to weaker cost controls.


Subject(s)
Endovascular Procedures/economics , Health Care Costs , Insurance, Health/economics , Intracranial Aneurysm/economics , Intracranial Aneurysm/surgery , Medicare/economics , Adult , Aged , Aged, 80 and over , Female , Humans , Insurance Coverage/economics , Intracranial Aneurysm/epidemiology , Length of Stay/economics , Male , Middle Aged , Retrospective Studies , Treatment Outcome , United States/epidemiology
10.
J Neurosurg Pediatr ; 27(2): 139-144, 2020 Dec 04.
Article in English | MEDLINE | ID: mdl-33276337

ABSTRACT

OBJECTIVE: The authors' previously published work validated the Chiari Health Index for Pediatrics (CHIP), a new instrument for measuring health-related quality of life (HRQOL) for pediatric Chiari malformation type I (CM-I) patients. In this study, the authors further evaluated the CHIP to assess HRQOL changes over time and correlate changes in HRQOL to changes in symptomatology and radiological factors in CM-I patients who undergo surgical intervention. Strong HRQOL evaluation instruments are currently lacking for pediatric CM-I patients, creating the need for a standardized HRQOL instrument for this patient population. This study serves as the first analysis of the CHIP instrument's effectiveness in measuring short-term HRQOL changes in pediatric CM-I patients and can be a useful tool in future CM-I HRQOL studies. METHODS: The authors evaluated prospectively collected CHIP scores and clinical factors of surgical intervention in patients younger than 18 years. To be included, patients completed a baseline CHIP captured during the preoperative visit, and at least 1 follow-up CHIP administered postoperatively. CHIP has 2 domains (physical and psychosocial) comprising 4 components, the 3 physical components of pain frequency, pain severity, and nonpain symptoms, and a single psychosocial component. Each CHIP category is scored on a scale, with 0 indicating absent and 1 indicating present, with higher scores indicating better HRQOL. Wilcoxon paired tests, Spearman correlations, and linear regression models were used to evaluate and correlate HRQOL, symptomatology, and radiographic factors. RESULTS: Sixty-three patients made up the analysis cohort (92% Caucasian, 52% female, mean age 11.8 years, average follow-up time 15.4 months). Dural augmentation was performed in 92% of patients. Of the 63 patients, 48 reported preoperative symptoms and 42 had a preoperative syrinx. From baseline, overall CHIP scores significantly improved over time (from 0.71 to 0.78, p < 0.001). Significant improvement in CHIP scores was seen in patients presenting at baseline with neck/back pain (p = 0.015) and headaches (p < 0.001) and in patients with extremity numbness trending at p = 0.064. Patients with syringomyelia were found to have improvement in CHIP scores over time (0.75 to 0.82, p < 0.001), as well as significant improvement in all 4 components. Additionally, improved CHIP scores were found to be significantly associated with age in patients with cervical (p = 0.009) or thoracic (p = 0.011) syrinxes. CONCLUSIONS: The study data show that the CHIP is an effective instrument for measuring HRQOL over time. Additionally, the CHIP was found to be significantly correlated to changes in symptomatology, a finding indicating that this instrument is a clinically valuable tool for the management of CM-I.


Subject(s)
Arnold-Chiari Malformation/psychology , Arnold-Chiari Malformation/surgery , Neurosurgical Procedures/methods , Neurosurgical Procedures/psychology , Adolescent , Child , Female , Follow-Up Studies , Health Status , Humans , Male , Pain Measurement , Postoperative Period , Predictive Value of Tests , Quality of Life , Retrospective Studies , Treatment Outcome , Young Adult
11.
J Neurosurg Pediatr ; 27(1): 16-22, 2020 Oct 09.
Article in English | MEDLINE | ID: mdl-33035994

ABSTRACT

OBJECTIVE: Parent or guardian involvement is implicit in the care of pediatric patients with hydrocephalus. Some parents and guardians are more engaged than others. The relationship between socioeconomic status (SES), the level of parental concern about their child's hydrocephalus management and future, and overall health status has not been clearly delineated. In this study, the authors sought to clarify this connection using hydrocephalus patient-reported health outcomes. METHODS: This cross-sectional study included children with surgically managed hydrocephalus whose parent or guardian completed the validated Hydrocephalus Outcome Questionnaire (HOQ) and Hydrocephalus Concern Questionnaire for parents (HCQ-P) on a return visit to the pediatric neurosurgery clinic at Vanderbilt University Medical Center between 2016 and 2018. Patients were excluded if the questionnaires were not completed in full. The calculated Overall Health Score (OHS) was used to represent the child's global physical, emotional, cognitive, and social health. The HCQ-P was used to assess parental concern about their child. Type of insurance was a proxy for SES. RESULTS: The HOQ and HCQ-P were administered and completed in full by 170 patient families. In the cohort, 91% of patients (n = 155) had shunt-treated hydrocephalus, and the remaining patients had undergone endoscopic third ventriculostomy. The mean (± SD) patient age was 12 ± 4 years. Half of the patients were male (n = 90, 53%), and most were Caucasian (n = 134, 79%). One in four patients lived in single-parent homes or with a designated guardian (n = 45, 26%). Public insurance and self-pay accounted for 38% of patients (n = 64), while the remaining 62% had private or military insurance. In general, parents with higher concern about their child's medical condition indicated that their son or daughter had a higher OHS (χ2 = 17.07, p < 0.001). Patients in families with a lower SES did not have different OHSs from those with a higher SES (χ2 = 3.53, p = 0.06). However, parents with a lower SES were more worried about management of their child's hydrocephalus and their child's future success (χ2 = 11.49, p < 0.001). In general, parents were not preoccupied with one particular aspect of their child's hydrocephalus management. CONCLUSIONS: More engaged parents, regardless of their family's SES, reported a better OHS for their child. Parents with public or self-paid insurance were more likely to report higher concern about their child's hydrocephalus and future, but this was not associated with a difference in their child's current health status.


Subject(s)
Disease Management , Health Surveys , Hydrocephalus/psychology , Hydrocephalus/therapy , Parents/psychology , Social Class , Adolescent , Child , Cross-Sectional Studies , Female , Health Status , Humans , Hydrocephalus/economics , Male , Prospective Studies , Retrospective Studies
12.
J Neurosurg Pediatr ; 26(3): 262-268, 2020 May 22.
Article in English | MEDLINE | ID: mdl-32442974

ABSTRACT

OBJECTIVE: The authors aimed to determine whether the Chiari Severity Index (CSI), and other clinical variables, can be used as a predictor of postoperative outcomes for Chiari type I malformation (CM-I) using the modified Chicago Chiari Outcome Scale (mCCOS) as the postoperative measure. METHODS: The cohort included patients 18 years of age and younger who were treated for CM-I between 2010 and 2015 who had at least 12 months of clinical and radiographic follow-up. CSI grades were assigned using preoperative clinical and neuroimaging data. Clinical, radiographic, and operative data were obtained from medical records. Kruskal-Wallis tests and Spearman correlations were conducted to assess for differences among CSI grades. Linear and ordinal regressions were conducted to evaluate predictors of the mCCOS and its components. Statistical significance was set a priori at p < 0.05. RESULTS: A total of 65 patients were included in the final cohort. The average age at the time of surgery and the mean mCCOS score were 9.8 ± 4.9 years and 10.4 ± 1.4, respectively. There were no significant differences in the mean mCCOS scores or CSI grades. Pre- and postoperative syrinx sizes were similar across the total patient cohort with median sizes of 7.4 and 3.7 mm, respectively. After controlling for age at the time of surgery, whether duraplasty and/or arachnoid dissection was performed, CSI preoperative score did not predict postoperative mCCOS score. No clinical variable could predict total mCCOS score. When the mCCOS was broken down into 3 subcomponents (pain, non-pain, and complications), only one relationship was identified. Those patients who presented with no headache had a statistically significant decrease in their pain (neck pain, shoulder pain, or dysesthesia in the upper extremities) as measured by the pain component of the mCCOS (χ2 [2, n = 20] = 6.43, p = 0.04). All other preclinical predictors, including CSI grades, were nonsignificant in demonstrating correlations to the mCCOS subcomponents. CONCLUSIONS: CSI grade was not found to be a marker of surgical outcome as measured by the mCCOS in this study. There were no correlations between the clinical variables and covariates investigated with the mCCOS. The lack of variation in mCCOS scores across this cohort may suggest that the mCCOS is not adequate for detecting differences in postsurgical outcomes. Further investigation is warranted to make this determination.

13.
J Neurosurg Pediatr ; 26(4): 415-420, 2020 May 29.
Article in English | MEDLINE | ID: mdl-32470930

ABSTRACT

OBJECTIVE: No study has established a relationship between cranial deformations and demographic factors. While the connection between the Back to Sleep campaign and cranial deformation has been outlined, considerations toward cultural or anthropological differences should also be investigated. METHODS: The authors conducted a retrospective review of 1499 patients (age range 2 months to less than 19 years) who presented for possible trauma in 2018 and had a negative CT scan. The cranial vault asymmetry index (CVAI) and cranial index (CI) were used to evaluate potential cranial deformations. The cohort was evaluated for differences between sex, race, and ethnicity among 1) all patients and 2) patients within the clinical treatment window (2-24 months of age). Patients categorized as "other" and those for whom data were missing were excluded from analysis. RESULTS: In the CVAI cohort with available data (n = 1499, although data were missing for each variable), 800 (56.7%) of 1411 patients were male, 1024 (79%) of 1304 patients were Caucasian, 253 (19.4%) of 1304 patients were African American, and 127 (10.3%) of 1236 patients were of Hispanic/Latin American descent. The mean CVAI values were significantly different between sex (p < 0.001) and race (p < 0.001). However, only race was associated with differences in positional posterior plagiocephaly (PPP) diagnosis (p < 0.001). There was no significant difference in CVAI measurements for ethnicity (p = 0.968). Of the 520 patients in the treatment window cohort, 307 (59%) were male. Of the 421 patients with data for race, 334 were Caucasian and 80 were African American; 47 of the 483 patients with ethnicity data were of Hispanic/Latin American descent. There were no differences between mean CVAI values for sex (p = 0.404) or ethnicity (p = 0.600). There were significant differences between the mean CVAI values for Caucasian and African American patients (p < 0.001) and rate of PPP diagnosis (p = 0.02). In the CI cohort with available data (n = 1429, although data were missing for each variable), 849 (56.8%) of 1494 patients were male, 1007 (67.4%) of 1283 were Caucasian, 248 (16.6%) of 1283 were African American, and 138 patients with ethnicity data (n = 1320) of Hispanic/Latin American descent. Within the clinical treatment window cohort with available data, 373 (59.2%) of 630 patients were male, 403 were Caucasian (81.9%), 84 were African American (17.1%), and 55 (10.5%) of 528 patients were of Hispanic/Latin American descent. The mean CI values were not significantly different between sexes (p = 0.450) in either cohort. However, there were significant differences between CI measurements for Caucasian and African American patients (p < 0.001) as well as patients of Hispanic/Latin American descent (p < 0.001) in both cohorts. CONCLUSIONS: The authors found no significant associations between cranial deformations and sex. However, significant differences exist between Caucasian and African American patients as well as patients with Hispanic/Latin American heritage. These findings suggest cultural or anthropological influences on defining skull deformations. Further investigation into the factors contributing to these differences should be undertaken.

14.
J Neurosurg Pediatr ; 26(2): 193-199, 2020 Apr 24.
Article in English | MEDLINE | ID: mdl-32330878

ABSTRACT

OBJECTIVE: The aim of this study was to determine the timeline of syrinx regression and to identify factors mitigating syrinx resolution in pediatric patients with Chiari malformation type I (CM-I) undergoing posterior fossa decompression (PFD). METHODS: The authors conducted a retrospective review of records from pediatric patients (< 18 years old) undergoing PFD for the treatment of CM-I/syringomyelia (SM) between 1998 and 2015. Patient demographic, clinical, radiological, and surgical variables were collected and analyzed. Radiological information was reviewed at 4 time points: 1) pre-PFD, 2) within 6 months post-PFD, 3) within 12 months post-PFD, and 4) at maximum available follow-up. Syrinx regression was defined as ≥ 50% decrease in the maximal anteroposterior syrinx diameter (MSD). The time to syrinx regression was determined using Kaplan-Meier analysis. Multivariate analysis was conducted using a Cox proportional hazards model to determine the association between preoperative, clinical, and surgery-related factors and syrinx regression. RESULTS: The authors identified 85 patients with CM-I/SM who underwent PFD. Within 3 months post-PFD, the mean MSD regressed from 8.1 ± 3.4 mm (preoperatively) to 5.6 ± 2.9 mm within 3 months post-PFD. Seventy patients (82.4%) achieved ≥ 50% regression in MSD. The median time to ≥ 50% regression in MSD was 8 months (95% CI 4.2-11.8 months). Using a risk-adjusted multivariable Cox proportional hazards model, the patients who underwent tonsil coagulation (n = 20) had a higher likelihood of achieving ≥ 50% syrinx regression in a shorter time (HR 2.86, 95% CI 1.2-6.9; p = 0.02). Thirty-six (75%) of 45 patients had improvement in headache at 2.9 months (IQR 1.5-4.4 months). CONCLUSIONS: The maximum reduction in syrinx size can be expected within 3 months after PFD for patients with CM-I and a syrinx; however, the syringes continue to regress over time. Tonsil coagulation was associated with early syrinx regression in this cohort. However, the role of surgical maneuvers such as tonsil coagulation and arachnoid veil identification and sectioning in the overall role of CM-I surgery remains unclear.

15.
Ann Noninvasive Electrocardiol ; 25(6): e12753, 2020 11.
Article in English | MEDLINE | ID: mdl-32198798

ABSTRACT

Patients with epilepsy suffer from a higher mortality rate than the general population, a portion of which is not due to epilepsy itself or comorbid conditions. Sudden unexpected death in epilepsy (SUDEP) is a common but poorly understood cause of death in patients with intractable epilepsy and often afflicts younger patients. The pathophysiology of SUDEP is poorly defined but does not appear to be related to prolonged seizure activity or resultant injury. Interestingly, a subset of patients with confirmed long QT syndrome (LQTS) present with a seizure phenotype and may have concurrent epilepsy. In this case, we present a patient who initially presented with a seizure phenotype. Further workup captured PMVT on an outpatient event monitor, and the patient was subsequently diagnosed with LQTS1. A substantial number of patients with LQTS initially present with a seizure phenotype. These patients may represent a subset of SUDEP cases resulting from ventricular arrhythmias. Appropriate suspicion for ventricular arrhythmias is necessary for proper arrhythmia evaluation and management in patients presenting with epilepsy.


Subject(s)
Death, Sudden , Electrocardiography/methods , Epilepsy/complications , Long QT Syndrome/complications , Long QT Syndrome/diagnosis , Adult , Epilepsy/physiopathology , Fatal Outcome , Female , Humans , Long QT Syndrome/physiopathology , Phenotype
16.
J Neurosurg Pediatr ; : 1-5, 2020 Jan 31.
Article in English | MEDLINE | ID: mdl-32005011

ABSTRACT

OBJECTIVE: The authors sought to assess the prevalence and severity of positional posterior plagiocephaly (PPP) in the pediatric population at a tertiary care center. METHODS: The authors conducted a retrospective review of 1429 consecutive patients aged 2 months to 18 years who presented with head trauma and a negative CT scan in 2018. The cohort was stratified by age. The cranial vault asymmetry index (CVAI) was calculated at the superior orbital rim. Asymmetry was categorized according to the following CVAI scores: mild (3.5%-7%), moderate (7%-12%), and severe (> 12%). Patients were grouped by age to assess PPP at different stages of head development: group 1, 2-5 months; group 2, 6-11 months; group 3, 12-23 months; group 4: 2-4 years; group 5, 5-8 years; group 6, 9-12 years; and group 7, 13-18 years. Patients with a history of shunted hydrocephalus, craniosynostosis, skull surgery, or radiographic evidence of intracranial trauma were excluded. RESULTS: The overall cohort prevalence of PPP was 24.8% (354 patients). PPP prevalence was higher among younger patients from groups 1-3 (40.4%, 33.5%, and 0.8%, respectively). There was a continued decline in PPP by age in groups 4-7 (26.4%, 20%, 20%, and 10.8%, respectively). Mild cranial vault asymmetry was noted most often (78.0%, 276 patients), followed by moderate (19.5%, 69 patients) and severe (2.5%, 9 patients). There were no patients in group 6 or 7 with severe PPP. CONCLUSIONS: PPP is prevalent in pediatric populations and most commonly presents as a case of mild asymmetry. Although there was an overall decline of PPP prevalence with increasing age, moderate asymmetry was seen in all age groups. No patients in the cohort had severe asymmetry that persisted into adolescence.

17.
Childs Nerv Syst ; 36(5): 1017-1024, 2020 05.
Article in English | MEDLINE | ID: mdl-31781913

ABSTRACT

PURPOSE: This study sought to determine the previously undescribed cytologic and metabolic alterations that accompany endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC). METHODS: Cerebrospinal fluid (CSF) samples were collected from infant patients with hydrocephalus at the time of index ETV/CPC and again at each reintervention for persistent hydrocephalus. Basic CSF parameters, including glucose, protein, and cell counts, were documented. A multivariable regression model, incorporating known predictors of ETV/CPC outcome, was constructed for each parameter to inform time-dependent normative values. RESULTS: A total of 187 infants were treated via ETV/CPC for hydrocephalus; initial laboratory values were available for 164 patients. Etiology of hydrocephalus included myelomeningocele (53, 32%), intraventricular hemorrhage of prematurity (43, 26%), aqueductal stenosis (24, 15%), and others (44, 27%). CSF parameters did not differ significantly with age or etiology. Glucose levels initially drop below population average (36 to 32 mg/dL) post-operatively before slowly rising to normal levels (42 mg/dL) by 3 months. Dramatically elevated protein levels post-ETV/CPC (baseline of 59 mg/dL up to roughly 200 mg/dL at 1 month) also normalized over 3 months. No significant changes were appreciated in WBC. RBC counts were very elevated following ETV/CPC and quickly declined over the subsequent month. CONCLUSION: CSF glucose and protein deviate significantly from normal ranges following ETV/CPC before normalizing over 3 months. High RBC values immediately post-ETV/CPC decline rapidly. Age at time of procedure and etiology have little influence on common clinical CSF laboratory parameters. Of note, the retrospective study design necessitates ETV/CPC failure, which could introduce bias in the results.


Subject(s)
Hydrocephalus , Neuroendoscopy , Third Ventricle , Cautery , Choroid Plexus/surgery , Humans , Hydrocephalus/etiology , Hydrocephalus/surgery , Infant , Laboratories , Retrospective Studies , Tertiary Care Centers , Third Ventricle/surgery , Treatment Outcome , Ventriculostomy
18.
J Neurosurg Pediatr ; : 1-7, 2019 Nov 29.
Article in English | MEDLINE | ID: mdl-31783365

ABSTRACT

OBJECTIVE: The authors aimed to evaluate clinical, radiological, and surgical factors associated with posterior fossa tumor resection (PFTR)-related outcomes, including postoperative complications related to dural augmentation (CSF leak and wound infection), persistent hydrocephalus ultimately requiring permanent CSF diversion after PFTR, and 90-day readmission rate. METHODS: Pediatric patients (0-17 years old) undergoing PFTR between 2000 and 2016 at Monroe Carell Jr. Children's Hospital of Vanderbilt University were retrospectively reviewed. Descriptive statistics included the Wilcoxon signed-rank test to compare means that were nonnormally distributed and the chi-square test for categorical variables. Variables that were nominally associated (p < 0.05) with each outcome by univariate analysis were included as covariates in multivariate linear regression models. Statistical significance was set a priori at p < 0.05. RESULTS: The cohort consisted of 186 patients with a median age at surgery of 6.62 years (range 3.37-11.78 years), 55% male, 83% Caucasian, and average length of follow-up of 3.87 ± 0.25 years. By multivariate logistic regression, the variables primary dural closure (PDC; odds ratio [OR] 8.33, 95% confidence interval [CI] 1.07-100, p = 0.04), pseudomeningocele (OR 7.43, 95% CI 2.23-23.76, p = 0.0007), and hydrocephalus ultimately requiring permanent CSF diversion within 90 days of PFTR (OR 9.25, 95% CI 2.74-31.2, p = 0.0003) were independently associated with CSF leak. PDC versus graft dural closure (GDC; 35% vs 7%, OR 5.88, 95% CI 2.94-50.0, p = 0.03) and hydrocephalus ultimately requiring permanent CSF diversion (OR 3.30, 95% CI 1.07-10.19, p = 0.0007) were associated with wound infection requiring surgical debridement. By multivariate logistic regression, GDC versus PDC (23% vs 37%, OR 0.13, 95% CI 0.02-0.87, p = 0.04) was associated with persistent hydrocephalus ultimately requiring permanent CSF diversion, whereas pre- or post-PFTR ventricular size, placement of peri- or intraoperative extraventricular drain (EVD), and radiation therapy were not. Furthermore, the addition of perioperative EVD placement and dural closure method to a previously validated predictive model of post-PFTR hydrocephalus improved its performance from area under the receiver operating characteristic curve of 0.69 to 0.74. Lastly, the authors found that autologous (vs synthetic) grafts may be protective against persistent hydrocephalus (p = 0.02), but not CSF leak, pseudomeningocele, or wound infection. CONCLUSIONS: These results suggest that GDC, independent of potential confounding factors, may be protective against CSF leak, wound infection, and hydrocephalus in patients undergoing PFTR. Additional studies are warranted to further evaluate clinical and surgical factors impacting PFTR-associated complications.

19.
J Neurosurg Pediatr ; 24(2): 145-152, 2019 May 31.
Article in English | MEDLINE | ID: mdl-31151096

ABSTRACT

OBJECTIVE: Open and endoscope-assisted repair are surgical options for sagittal craniosynostosis, with limited research evaluating each technique's immediate and long-term costs. This study investigates the cost-effectiveness of open and endoscope-assisted repair for single, sagittal suture craniosynostosis. METHODS: The authors performed a retrospective cohort study of patients undergoing single, sagittal suture craniosynostosis repair (open in 17 cases, endoscope-assisted in 16) at less than 1 year of age at Monroe Carell Jr. Children's Hospital at Vanderbilt (MCJCHV) between August 2015 and August 2017. Follow-up data were collected/analyzed for 1 year after discharge. Surgical and follow-up costs were derived by merging MCJCHV financial data with each patient's electronic medical record (EMR) and were adjusted for inflation using the healthcare Producer Price Index. Proxy helmet costs were derived from third-party out-of-pocket helmet prices. To account for variable costs and probabilities, overall costs were calculated using TreeAge tree diagram software. RESULTS: Open repair occurred in older patients (mean age 5.69 vs 2.96 months, p < 0.001) and required more operating room time (median 203 vs 145 minutes, p < 0.001), more ICU days (median 3 vs 1 day, p < 0.001), more hospital days (median 4 vs 1 day, p < 0.001), and more frequently required transfusion (88% vs 6% of cases). Compared to patients who underwent open surgery, patients who underwent endoscopically assisted surgery more often required postoperative orthotic helmets (100% vs 6%), had a similar number of follow-up clinic visits (median 3 vs 3 visits, p = 0.487) and CT scans (median 3 vs 2 scans), and fewer emergency department visits (median 1 vs 3 visits). The TreeAge diagram showed that, overall, open repair was 73% more expensive than endoscope-assisted repair ($31,314.10 vs $18,081.47). Sensitivity analysis identified surgical/hospital costs for open repair (mean $30,475, SEM $547) versus endoscope-assisted repair (mean $13,746, SEM $833) (p < 0.001) as the most important determinants of overall cost. Two-way sensitivity analysis comparing initial surgical/hospital costs confirmed that open repair remains significantly more expensive under even worst-case initial repair scenarios ($3254.81 minimum difference). No major surgical complications or surgical revisions occurred in either cohort. CONCLUSIONS: The results of this study suggest that endoscope-assisted craniosynostosis repair is significantly more cost-effective than open repair, based on markedly lower costs and similar outcomes, and that the difference in initial surgical/hospital costs far outweighs the difference in subsequent costs associated with helmet therapy and outpatient management, although independent replication in a multicenter study is needed for confirmation due to practice and cost variation across institutions. Longer-term results will also be needed to examine whether cost differences are maintained.

20.
J Neurosurg Pediatr ; 24(2): 159-165, 2019 May 24.
Article in English | MEDLINE | ID: mdl-31125958

ABSTRACT

OBJECTIVE: Little is understood about the role that health disparities play in the treatment and management of brain tumors in children. The purpose of this study was to determine if health disparities impact the timing of initial and follow-up care of patients, as well as overall survival. METHODS: The authors conducted a retrospective study of pediatric patients (< 18 years of age) previously diagnosed with, and initially treated for, a primary CNS tumor between 2005 and 2012 at Monroe Carell Jr. Children's Hospital at Vanderbilt. Primary outcomes included time from symptom presentation to initial neurosurgery consultation and percentage of missed follow-up visits for ancillary or core services (defined as no-show visits). Core services were defined as healthcare interactions directly involved with CNS tumor management, whereas ancillary services were appointments that might be related to overall care of the patient but not directly focused on treatment of the tumor. Statistical analysis included Pearson's chi-square test, nonparametric univariable tests, and multivariable linear regression. Statistical significance was set a priori at p < 0.05. RESULTS: The analysis included 198 patients. The median time from symptom onset to initial presentation was 30.0 days. A mean of 7.45% of all core visits were missed. When comparing African American and Caucasian patients, there was no significant difference in age at diagnosis, timing of initial symptoms, or tumor grade. African American patients missed significantly more core visits than Caucasian patients (p = 0.007); this became even more significant when controlling for other factors in the multivariable analysis (p < 0.001). African American patients were more likely to have public insurance, while Caucasian patients were more likely to have private insurance (p = 0.025). When evaluating survival, no health disparities were identified. CONCLUSIONS: No significant health disparities were identified when evaluating the timing of presentation and survival. A racial disparity was noted when evaluating missed follow-up visits. Future work should focus on identifying reasons for differences and whether social determinants of health affect other aspects of treatment.

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